Gatlinburg Conference 40 Annual On Research and Theory in Intellectual & Developmental Disabilities

40th Annual
Gatlinburg Conference
On Research and Theory in Intellectual & Developmental Disabilities
The Loews Annapolis • Annapolis • Maryland
March 7-9, 2007
This volume contains abstracts for invited talks, individual and symposia papers, and poster presentations at the
40th Annual Gatlinburg Conference on Research and Theory in Intellectual and Developmental Disabilities,
held March 7-9, 2007, in Annapolis, Maryland.
Permission to quote or reprint any of these materials must be obtained from the author(s).
• EXECUTIVE COMMITTEE •
Leonard Abbeduto, Conference Co-Chair
University of Wisconsin–Madison
Jennifer Hill Karrer
University of Victoria
Don Bailey
University of North Carolina at Chapel Hill
Connie Kasari
University of California at Los Angeles
John Borkowski
Notre Dame University
Mark Lewis
University of Florida
Frances Conners
University of Alabama
William MacLean
University of Wyoming
Francis Crinella
University of California at Irvine
Marsha Mailick Seltzer, Conference Co-Chair
University of Wisconsin–Madison
Sharon Duffy
University of California at Riverside
William McIlvane
University of Massachusetts Medical School
Elisabeth Dykens
Vanderbilt University
Richard Serna
University of Massachusetts Medical School
Deborah Fidler
Colorado State University
Wayne Silverman
Johns Hopkins University School of Medicine
Frank Floyd
Georgia State University
Steve Warren
University of Kansas
Susan Hepburn
University of Colorado
• THEME COMMITTEE •
Frances Conners
University of Alabama
Jennifer Hill Karrer
University of Victoria
Elisabeth Dykens
Vanderbilt University
Wayne Silverman, Chair
Johns Hopkins University School of Medicine
Deborah Fidler
Colorado State University
• CONFERENCE COORDINATION •
Teresa Palumbo – Conference Coordinator
Waisman Center, University of Wisconsin-Madison
Ernie Colantonio, AV Coordinator
Waisman Center, University of Wisconsin-Madison
• CONFERENCE SUPPORT•
This conference is supported by grant number R13 HD38335 from the
National Institute of Child Health and Human Development.
Additional support provided through the generous donations of Brookes Publishing, Dr. Robert Littleton and Evergreen
Center, Inc., and Dr. Michael Cataldo and the Kennedy Krieger Institute.
1
Ta b l e o f C o n t e n t s
P R O G R A M AT A GLANCE ......................................................................................... 3
A W A R D R E C I P I E N T S ............................................................................................ 4
E X H I B I T O R S .............................................................................................................. 5
I N V I T E D P R E S E N T E R S....................................................................................... 6
D E T A I L E D C O N F E R E N C E P R O G R A M ........................................................ 8
WEDNESDAY P R E S E N T A T I O N S
MORNING
• Symposium 1 ................................................................................................................. 21
• Symposium 2 ................................................................................................................. 29
• Symposium 3 ................................................................................................................. 35
AFTERNOON
• Symposium 4 ................................................................................................................. 40
• Symposium 5 ................................................................................................................. 45
• Symposium 6 ................................................................................................................. 50
EVENING
• Poster Session 1 ............................................................................................................ 55
T H U R S D AY P R E S E N TAT I O N S
MORNING
• Symposium 7 ................................................................................................................. 82
• Symposium 8 ................................................................................................................. 87
• Symposium 9 ................................................................................................................. 92
AFTERNOON
• Paper Session 1 ............................................................................................................. 98
• Paper Session 2 ............................................................................................................. 105
• Paper Session 3 ............................................................................................................. 112
EVENING
• Poster Session 2 ............................................................................................................ 119
F R I D AY P R E S E N TAT I O N S
MORNING
• Symposium 10 ............................................................................................................... 145
• Symposium 11 ............................................................................................................... 151
• Symposium 12 ............................................................................................................... 156
A U T H O R I N D E X ................................................................................................. 162
2
2007 Gatlinburg Conference
Program at a Glance
TUESDAY, MARCH 6, 2007
5:00 - 7:00 P.M.
REGISTRATION
LOBBY FOYER
WEDNESDAY, MARCH 7, 2007
7:30 - 8:45 A.M.
REGISTRATION
8:45 - 10:15 A.M.
SYMPOSIUM 2
SYMPOSIUM 1
SYMPOSIUM 3
BALLROOM B
BALLROOM A
BALLROOM C
Opening Remarks: Marsha Mailick Seltzer, PhD & Len Abbeduto, PhD
REGATTA BALLROOM ABC
Plenary Session: Stephanie Sherman, PhD
REGATTA BALLROOM ABC
10:30 - 10:45 A.M.
10:45 A.M. - 12:15 P.M.
ATRIUM
1:15 - 2:45 P.M.
SYMPOSIUM 4
BALLROOM A
4:45 P.M. - 6:00 P.M.
NIH Workshop: The NIH Organization, Programs, & Grant Review
REGATTA BALLROOM ABC
6:30 P.M. - 8:30 P.M.
Poster Session 1
SYMPOSIUM 5
BALLROOM B
SYMPOSIUM 6
BALLROOM C
ATRIUM
THURSDAY, MARCH 8, 2007
SYMPOSIUM 8
BALLROOM B
8:45 - 10:15 A.M.
SYMPOSIUM 7
BALLROOM A
10:30 A.M. - 12:00 P.M.
Plenary Session: Ira Lott, MD
REGATTA BALLROOM ABC
1:15 - 2:45 P.M.
Plenary Session: Jennifer Wishart, PhD
REGATTA BALLROOM ABC
3:00 - 5:00 P.M.
PAPER SESSION 1
BALLROOM A
5:15 P.M. - 6:15 P.M.
PAPER SESSION 2
BALLROOM B
Update on NIH Task Force on Down Syndrome
6:30 P.M. - 8:30 P.M.
Poster Session 2
SYMPOSIUM 9
BALLROOM C
PAPER SESSION 3
BALLROOM C
REGATTA BALLROOM ABC
ATRIUM
FRIDAY, MARCH 9, 2007
8:45 - 10:15 A.M.
SYMPOSIUM 10
BALLROOM A
SYMPOSIUM 11
BALLROOM B
SYMPOSIUM 12
BALLROOM C
10:30 A.M. - 12:00 P.M.
Plenary Session: Roger Reeves, PhD
REGATTA BALLROOM ABC
12:00 P.M.
Closing Remarks: Marsha Mailick Seltzer, PhD & Len Abbeduto, PhD
REGATTA BALLROOM ABC
3
Award Recipients
David Zeaman Student Award
R. Micheal Barker
Georgia State University
Athena Hayes
University of Wyoming
Joanne Kersh
Boston College
Sara Kover
University of Wisconsin-Madison
Tracey Lloyd
University of Wales, Bangor
Caitlin McMahon
Vanderbilt University
Irene Ngai
Georgia State University
Ashlyn Smith
Georgia State University
Theodore Tjossem
Po s t - Do c t o r a l Fe l l o w Aw a rd
Lara Head
University of Wisconsin-Madison
Nancy Raitano Lee
University of Colorado at Denver
4
Exhibitors
• The Gatlinburg Conference especially thanks Brookes Publishing Co. for its generous
sponsorship of the Poster Session 2 reception and a student travel award •
Brookes Publishing Co.
P.O. Box 10624
Baltimore, MD 21285-0624
Phone: 1-800-638-3775 • Fax: 410-337-8539
Email: [email protected]
www.brookespublishing.com
Stop by the booth at 6:00 P.M. Wednesday, March 7 and at 5:00 p.m. on Thursday, March 8
for the FREE student book drawings
Blackwell Publishing
350 Main Street
Malden MA 02149
Phone: 781-388-8200
Fax: 781-388-8210
www.blackwellpublishing.com
E-mail:
[email protected]
Caring Technologies Inc
1423 W. Franklin St.
Boise, ID 83702
Phone/Fax 1-888-355-7161
Ron Oberleitner CEO
www.caringtechnologies.com
E-mail: [email protected]
• THANK YOU TO OUR 2007 EXHIBITORS •
5
Invited Speakers
Ira Lott, MD
Thursday, March 8, 2007
10:30 A.M.–12:00 P.M.
“Down Syndrome and Alzheimer’s Disease: Clues from Neuroimaging”
Regatta Ballroom
Individuals with Down syndrome (DS) are at risk for developing Alzheimer disease after the age of
40 years. It is not clear why some individuals display cognitive decline and others do not even in the 6th
or 7th decades of life. Functional brain imaging may help to elucidate the pathogenesis of AD in DS.
In non-demented subjects with DS, we have reported an increased glucose metabolic rate by PET
scanning in the same areas of the brain (inferior temporal lobe/entorhinal cortex) where patients with
AD in the general population show decreased activity. This suggests that a neuronal compensatory
response rate may precede the development of dementia in DS. Evidence supporting this hypothesis is
seen through immunohistchemical studies in which we have shown that in middle age adults with DS,
prior to entorhinal neuron loss, the earliest tau accumulation occurred in the outer molecular layer.
These events were followed at a later age, and associated with entorhinal neuron loss, by an increase in
GAP-43. Hilar neurons exhibiting a sprouting morphology were also noted. More recently, we have
studied the relationship between PET, gray matter assessed with voxel-based morphometry, and dementia
ratings in clinically non-demented individuals with DS. The strongest correlations to ratings of dementia
included the combination of higher GMR and decreased gray matter volume in parts of the temporal
cortex, including the parahippocampus/hippocampus, in the thalamus, caudate, and frontal lobe (BA
47).The combination of increased GMR overlapping with less gray matter in these areas may be consistent with a compensatory brain
response to an early stage of the disease process. Taken together with the clinical findings, PET studies in DS may aid in early detection
of dementia even before any tissue loss is detected by MRI scans.
Dr. Lott is the associate dean of clinical neurosciences, director of the Division of Child Neurology and professor of pediatrics and neurology
at the University of California, Irvine
Roger H. Reeves, PhD
Friday, March 9, 20067
10:30 A.M.–12:00 P.M.
Regatta Ballroom
“Therapy for Structural Anomalies in the Trisomic Brain”
Down syndrome (DS) is the most complex genetic challenge compatible with human survival.
Because the pathologies associated with DS are the products of elevated gene expression in every cell
throughout development, it has often been assumed to be too complex for meaningful intervention.
Recent basic science advances show promise for a meaningful approach to understand the basis for
pathogenesis in DS which will support the search for treatments. At least two approaches demonstrate
possibilities for overcoming fundamental aspects of the developmental/ageing effects on the CNS in
DS. We will review experimental approaches to define the complex genetic basis for variability in
cognitive phenotypes. Results from animal model studies in which treatment with a small molecule
can reverse structural anomalies in the brain will be presented.
Dr. Reeves is a professor in the Department of Physiology at the Johns Hopkins University School of
Medicine and Institute for Genetic Medicine.
6
Invited Speakers
Stephanie Sherman,PhD
Wednesday, March 7, 2007
10:45 A.M.–12:15 P.M.
Regatta Ballroom
"Trisomy 21: Causes and Consequences"
Human chromosome nondisjunction leads to an extraordinary frequency of aneuploidy. For the
past 15 years, we have investigated trisomy 21, the leading cause of Down syndrome (DS), as a model
to understand nondisjunction and its phenotype consequences. We have built an unprecedented resource of infants with DS and their parents, including biological samples, epidemiological and clinical
data. With respect to risk factors associated with nondisjunction, we have shown that altered recombination patterns along the nondisjoined chromosome are associated with maternal nondisjunction.
This association is observed for errors classified as occurring in meiosis I (MI), when recombination
takes place, and in meiosis II (MII), long after recombination is completed. This latter finding suggests
that so-called “MII” errors are initiated in MI. We have now shown that the position of susceptible
recombinants differ by the age of the oocyte. Intriguingly, we find that this maternal-age related pattern differs between MI and MII errors. For MI errors, the proportion of oocytes with susceptible
telomeric recombinants is highest among young mothers and decreases significantly in the oldest age
group. Consequently, we hypothesize that altered recombination is an initial susceptibility factor for
nondisjunction, sometimes followed by the perturbation of the meiotic process at a later time. This
second step may be age-independent (e.g., genetic or environmental factors) or age-dependent (e.g.,
accumulated environmental insults). In contrast, among MII errors, susceptible pericentromeric recombination occurs most often in the
older age group of oocytes compared with the younger age group. Our ultimate goal is to dissect that susceptibility for nondisjunction
into its primary liability factors to better determine potential treatment endpoints. With respect to phenotype consequences, our goal is
to identify genetic and environmental factors that increase the risk for Down syndrome-associated birth defects. We have begun by
examining congenital heart defects as a paradigm for the study of more complex traits, such as cognition and behavior. To begin, we have
established a multi-site project to enroll participants with Down syndrome and complete atrioventricular septal defects (AVSD) and
those without heart defects. We will conduct a genetic association case-control study to identify susceptibility genes for AVSD. We will
use our data from the epidemiological study to identify environmental risk factors and potential gene-environment interactions.
Dr. Sherman is a professor in the Department of Human Genetics and director of the Genetics and Molecular Biology Graduate Program at
Emory University School of Medicine.
Jennifer G. Wishart,PhD
Thursday, March 8, 2007
1:15 P.M.–2:45 P.M.
Regatta Ballroom
“Social Cognition In Children with Down Syndrome:
Current Evidence, Future Directions”
Down syndrome (DS) is one of the best known of all of the intellectually-disabling conditions and
yet surprisingly it is still possibly one of the least well understood. Most children with DS face major
obstacles in acquiring basic childhood skills and also usually face significant difficulties in forming and
maintaining peer friendships. Comparatively little empirical research directly addresses the origins of
these difficulties and indeed social understanding is commonly thought to be relatively ‘protected’ in
this syndrome. This talk aims to overview findings from recent evidence on socio-cognitive skills in
DS, assessing the possible developmental implications of any weaknesses in this area of functioning.
Data from our own most recent work will also be presented, including studies of emotion recognition
ability and of behaviors in collaborative learning contexts. Our overall aim has been to identify any
relative strengths and weaknesses in socio-cognitive understanding in children with DS, to investigate
how they make use of their interpersonal understanding in potential learning contexts, and to try to tie
what is known about social understanding and developmental trajectories in DS to what is known
about the underlying neurobiology of DS. Our findings suggest that motivational and socio-cognitive
deficits may add to the effects of general cognitive impairment in DS and contribute to the decline in
developmental rate characteristic of this syndrome. Wide variability in individual developmental profiles,
however, suggests that some of these psychologically-driven aspects of behavior may be open to facilitative
intervention, unlike many of the biologically-determined weaknesses in DS functioning.
Dr. Wishart is a professor of developmental disabilities in childhood at the Moray House School of Education, University of Edinburgh
7
2007 Gatlinburg Conference
W E D N E S D A Y
March 7, 2007
8:45 – 10:15 A.M.
• SYMPOSIUM 1 – Ballroom A
Early Risk for Developmental Delays Among
Children of First Time Mothers: Two Year Findings
from the Parenting for the First Time Project
Chair: John G. Borkowski, University of Notre Dame
Discussant: Craig Ramey, Georgetown University
The Parenting for the First Time Project: Design and
Methodology
Jennifer Burke Lefever1
Robin Gaines Lanzi2
Centers for the Prevention of Child Neglect3
1
University of Notre Dame
Georgetown University
3
Georgetown University, University of Alabama-Birmingham,
University of Kansas, University of Notre Dame, University of
Texas at Houston
2
Prenatal Cumulative Risk and Maternal Parenting
During Infancy
Kimberly S. Howard1
Judith J. Carta2
Kere P. Hughes3
Julie N. Schatz4
John G. Borkowski4
Centers for the Prevention of Child Neglect5
1
Teachers College, Columbia University
University of Kansas
3
Iowa State University
4
University of Notre Dame
5
Georgetown University, University of Alabama-Birmingham,
University of Kansas, University of Notre Dame, University of
Texas at Houston
2
Application of the E-RISK Index with Children with
Special Needs: A Reliable and Easy-to Administer
Screening Tool of Early Precursors of Likely
Neglectful Behaviors
Robin Gaines Lanzi1
Sharon Landesman Ramey1
Shannon Carothers1
Bette R. Keltner1
Centers for the Prevention of Neglect2
1
Georgetown University
Georgetown University, University of Alabama-Birmingham,
University of Kansas, University of Notre Dame, University of
Texas at Houston
2
The Longitudinal Impact of Maternal Warmth and
Hostility on Early Language Development:
Comparisons of Adolescent and Adult Mothers
Leann E. Smith1
Gitta Lubke2
Steve Warren3
Centers for the Prevention of Child Neglect4
8
1
University of Wisconsin-Madison
University of Notre Dame
3
University of Kansas
4
Georgetown University, University of Alabama-Birmingham,
University of Kansas, University of Notre Dame, University of
Texas at Houston
2
Prenatal Maltreatment Risk, Early Parenting
Behaviors, and Children’s Emergent Regulation
Julie Schatz1
John G. Borkowski1
Centers for the Prevention of Child Neglect2
1
University of Notre Dame
Georgetown University, University of Alabama-Birmingham,
University of Kansas, University of Notre Dame, University of
Texas at Houston
2
Obstacles to Service Receipt for Children with Early
Indicators of Developmental Delays
Carol E. Akai1
Leann E. Smith2
Lorraine V. Klerman3
Bette R. Keltner4
Kristi C. Guest5
Centers for the Prevention of Child Neglect6
1
University of Notre Dame
University of Wisconsin—Madison
3
Brandeis University
4
Georgetown University
5
University of Alabama at Birmingham
6
Georgetown University, University of Alabama-Birmingham,
University of Kansas, University of Notre Dame, University of
Texas at Houston
2
• SYMPOSIUM 2 – Ballroom B
Down Syndrome, Aging, and Alzheimer’s Disease:
Practical Insights from Prospective Studies
Chair:Wayne Silverman, Kennedy Krieger Institute and
Johns Hopkins University School of Medicine
Elevated Plasma Amyloid β -Peptide Aβ42,Incident
Dementia and Mortality in Down Syndrome
Nicole Schupf1
Bindu P. Patel2
Deborah Pang2
Warren B. Zigman3
Pankaj D. Mehta3
Richard P. Mayeux2
Wayne Silverman4
1
Columbia University, G. H. Sergievsk Center
Columbia University Medical Center
3
NYS Institute for Research on Developmental Disabilities
4
Kennedy Krieger Institute and Johns Hopkins School of
Medicine
2
2007 Gatlinburg Conference
W E D N E S D A Y
March 7, 2007
Apolipoprotein ε4 is Associated with Increased
Mortality Risk in Nondemented Adults with Down
Syndrome
Warren B. Zigman1
Edmund C. Jenkins1
Benjamin Tycko2
Nicole Schupf1,3
Wayne Silverman4
1
New York State Institute for Basic Research in
Developmental Disabilities
2
The Taub Institute for Research on Alzheimer’s Disease and
the Aging Brain, Columbia University College of Physicians
and Surgeons
3
Columbia University, G. H. Sergievski Center
4
Kennedy Krieger Institute and Johns Hopkins University
School of Medicine
“Mild Cognitive Impairment” and Behavioral
Concerns in Adults with Down Syndrome
Tiina K. Urv1
Sharon Krinsky-McHale2
Warren Zigman2
Wayne Silverman3
Sibling Relationships and Psychological Well-Being
in Adults with and Without Siblings with Intellectual
Disabilities
Richard Hastings1
Mairead Doody1
Sarah O’Neill2
Ian Grey2
1
2
University of Wales Bangor
Trinity College Dublin
Parent – Sibling Communication in Families of
Children with Autism, Down Syndrome, and Sickle
Cell Disease
J. Carolyn Graff
University of Tennessee Health Science Center
10:30 – 10:45 A.M.
• OPENING REMARKS–
REGATTA BALLROOM
Marsha Mailick Seltzer, PhD & Len Abbeduto, PhD
Waisman Center, University of Wisconsin-Madison
1
University of Massachusetts Medical School–Shriver Center
New York State Institute for Basic Research in
Developmental Disabilities
3
Kennedy Krieger Institute and Johns Hopkins University
School of Medicine
2
Are Cholinesterase Inhibitors Effective for Adults
with Down Syndrome?
Wayne Silverman1
Warren Zigman2
Nicole Schupf2,3
Tiina Urv4
Robert Ryan2
1
Kennedy Krieger Institute and Johns Hopkins University
School of Medicine
2
New York State Institute for Basic Research in
Developmental Disabilities
3
Columbia University, G. H. Sergievski Center
4
University of Massachusetts Medical School–Shriver Center
• SYMPOSIUM 3 – Ballroom C
Siblings of Individuals with Developmental
Disabilities Across the Life Span
Chair: Ann Kaiser, Vanderbilt University
Discussant: Gael Orsmond, Boston University
Adult Siblings of Persons with Disabilities:
Findings from a National Survey
Richard C. Urbano
Robert M. Hodapp
10:45 A.M. – 12:15 P.M.
• PLENARY SESSION–REGATTA BALLROOM
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
"Trisomy 21: Causes and Consequences"
Stephanie Sherman, PhD
Professor of human genetics and director of the Genetics and
Molecular Biology Graduate Program, Emory University
School of Medicine.
1:15 P.M. – 2:45 P.M.
• SYMPOSIUM 4 – Ballroom A
Asian Families of Children with Developmental
Disabilities
Chair: Jin Y. Shin, Hofstra University
Discussant: Laraine M. Glidden,Georgetown University
Agreement on Childhood Disability between
Parents and Teachers in Vietnam
Jin Y. Shin1,2
Nguyen Viet Nhan 3
Stavros S. Valenti1
Michel Flory 2
1
Department of Psychology, Hofstra University
2
Infant Development, New York State Institute for Basic
Research in Developmental Disabilities
3
Hue Medical College, Vietnam
Vanderbilt University
Symposium 4 continued on next page
9
2007 Gatlinburg Conference
W E D N E S D A Y
March 7, 2007
A Comparison of Perspectives on Sibling
Relationships Involving an Individual with Special
Needs
Sun Young Ryu
Department of Health and Behavioral Studies, Teachers
College, Columbia University
Parent Language Input and Its Impact on the
Engagement of Children with Autism: The Chinese
Perspective
Peishi Wang
Graduate Program in Special Education, Department of
Educational and Community Programs, Queens College,
City University of New York
• SYMPOSIUM 5 – Ballroom B
Children with Down Syndrome: Parent and Child
Supports
Chair: Mary Ann Romski, Georgia State University
Discussant: Nancy Brady, University of Kansas
Parent-Child Communication at Home in Preschool
Children with Down Syndrome and Autism
Spectrum Disorders
Ann P. Kaiser1
Terry B. Hancock1
Prathibha Vijay1
1
Vanderbilt University
Coping with Frustration in Children with Down
Syndrome
Laudan B. Jahromi1
Amanda Gulsrud2
Connie Kasari2
1
Arizona State University
2
University of California, Los Angeles
Examining Parental Stress and Parental Perceptions
of Toddler’s Communication Development
Following Language Intervention: A Comparison
between Children with and without Down Syndrome
Ashlyn L. Smith1
Mary Ann Romski1
1
Georgia State University
• SYMPOSIUM 6 – Ballroom C
Down Syndrome: Gene Expression and Beyond
Chair: Benjamin Tycko, Institute for Cancer Genetics,
Herbert Irving Cancer Research Center
Discussant: Nicole Schupf, Taub Institute for Research
on Alzheimer’s Disease and the Aging Brain, Columbia
University Medical CenterInstitute for Basic Research in
Developmental Disabilities
10
Function of Chromosome 21 Gene Products
Investigated in BAC-Transgenic Mice
Luzhou Xing
Martha Salas
Chyuan-Sheng Lin
Ning Kon
Muyang Li
Wei Gu
Warren Zigman
Wayne Silverman
Benjamin Tycko
Department of Pathology, Taub Institute and Institute for
Cancer Genetics, Columbia University Medical Center
Department of Psychology, New York State Institute for Basic
Research
Department of Behavioral Psychology, Kennedy Krieger
Institute
Role of PIP2 Phosphatase Synaptojanin 1 in Down
Syndrome-Linked Brain Dysfunction
Sergey V. Voronov
Samuel G. Frere
Silvia Giovedi
Elizabeth A. Pollina
Cecilia Schmidt
Laurent Cimasoni
Christelle Borel
Markus R. Wenk
Stylianos E. Antonarakis
Katheleen Gardiner
Muriel T. Davisson
Ottavio Arancio
Pietro De Camilli
Gilbert Di Paolo
Taub Institute for Research on Alzheimer’s Disease and the
Aging Brain
Columbia University Medical Center
Consequences of Over-Expression of Mnb/Dyrk1A
in Down Syndrome
Jerzy Wegiel
Karol W. Dowjat
Wojciech Kaczmarski
Yu-Wen Hwang
Cheng-Xin Gong
Tatyana Adayev
Madhabi Barua
Izabela Kuchna
Krzysztof Nowicki
Jarek Wegiel
Humi Imaki
Pankaj Mehta
Arthur Dalton
Georgio Albertini
Symposium 6 continued on next page
2007 Gatlinburg Conference
W E D N E S D A Y
March 7, 2007
Ausma Rabe
Zhihou Liang
Fei Liu
Khalid Iqbal
Inge Grundke-Iqbal
Noriko Murakami
Naryan Ramakrishna
Wayne P. Silverman
New York State Institute for Basic Research in
Developmental Disabilities
Kennedy Krieger Institute, Johns Hopkins University School
of Medicine
4:45 P.M. – 6:00 P.M.
• PLENARY SESSION–REGATTA BALLROOM
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
NIH Workshop: The NIH Organization,
Programs, & Grant Review
6:30 P.M. – 8:30 P.M.
• POSTER SESSION 1– ATRIUM
1. Hyporesponsive Sensory Patterns in Young
Children with Autism,Developmental Delay, and
Typical Development
Grace T. Baranek
Margaret DeRamus
Linda R. Watson
Brian A. Boyd
Michele D. Poe
University of North Carolina at Chapel Hill
2. Intrauterine Growth Retardation and Executive
Function Performance at School Age
Richard C. Belser
Vicki Sudhalter
Judith M. Gardner
Bernard Z. Karmel
N.Y.S. Institute for Basic Research in Developmental
Disabilities
3. Persistence in Early Communication by Young
Children with Down Syndrome and Other
Developmental Disabilities
Nancy C. Brady
Kandace Fleming
4. Experiences of Parental Advocates in Self-help
Groups For Children with Autism
Irene Carter
University of Calgary
5. Early Learning Profile in Preschoolers with
Williams Syndrome: A Descriptive Study
Ashley Cole
David Most
Deborah Fidler
Amy Philofsky
Susan Hepbun
Colorado State University; University of Colorado Denver
Health Sciences Center
6. Predicting Marital Satisfaction from Initial
Maternal Depression and Burden of Care
Jenalee Coster
Brian M. Jobe
Laraine M. Glidden
St. Mary’s College of Maryland
7. Factors Predicting Mortality in a Residing with
Family Aging Sample of Adults with Mental
Retardation
Anna J. Esbensen
Marsha Mailick Seltzer
Jan S. Greenberg
Waisman Center, University of Wisconsin – Madison
8. Examining Differences between Autistic Children
with and without Mental Retardation
Marygrace Y. Kaiser
Vanessa Gonzalez
Jennifer S. Durocher
Michael Alessandri
Claudia Rojas
University of Miami
9. Linking Executive Function and Repetitive
Behaviors in Autism Spectrum Disorders
Harrison, B.1
Black, D.2
Wallace, G. 3
Della Rosa, A.1
Kenworthy, L.1
1
Children’s National Medical Center, Washington, D.C.
Boston University School of Medicine
3
National Institute of Mental Health
2
University of Kansas
Poster Session 1 continued on next page
11
2007 Gatlinburg Conference
W E D N E S D A Y
March 7, 2007
10. Restricted, Repetitive and Stereotyped
Behaviors, Interests, and Activities in Children with
Autism
Athena Hayes1
William MacLean, Jr.1
Susan Hepburn2
Sally Rogers3
1
University of Wyoming
University of Colorado Health Sciences Center
3
University of California-Davis
2
11. Enhancing Cognitive-Emotional Skills for
Children with Developmental Disabilities through
Computer-Based Training
Ingrid M. Hopkins
Margaret J. Bates
Shana B. Crowson
Rebekah K. Pearson
Fred J. Biasini
University of Alabama at Birmingham
12. Increased Special Health Care Needs in Children
with Severe Developmental Disabilities and
Behavioral Disorders: Implications for
Interdisciplinary Treatment of Problem Behavior
Craig H. Kennedy
A. Pablo Juárez
Angela Becker
Kathryn Greenslade
Mark T. Harvey
Clare Sullivan
Brenna Tally
Vanderbilt University
13. Teaching Money Skills to Children with
Intellectual Disabilities: Rapid Learning via
Constructed Response Matching-to-Sample
Procedures
Joanne B. Kledaras1
Dana Hurlbut1
William J. McIlvane2
William V. Dube2
1
2
Praxis, Inc.
UMMS Shriver Center
14. Receptive Language in Adolescents with Fragile
X Syndrome with or without Autism
S. T. Kover
L. Abbeduto
N. D. Giles
E. K. Richmond
P. Lewis
A. Weber
S. Schroeder
J. A. Anderson
A. Baron
Waisman Center, University of Wisconsin-Madison
15. Developmental Trajectories of Language Skills
in Toddlers and Adolescents with Down Syndrome
Nancy Raitano Lee1,2
Susan Hepburn1
Amy Philofsky1
Deborah Fidler3
Sally Rogers4
Bruce Pennington2
1
JFK Partners; University of Colorado at Denver & Health
Sciences Center
2
Department of Psychology, University of Denver
3
Department of Human Development & Family Studies,
Colorado State University
4
M.I.N.D. Institute, University of California – Davis
16. Mood and Anxiety in Mothers of Children with
FXS and Mothers of Children with Autism
Jean B. Mankowski1
Jane Roberts1
Donald Bailey Jr2
1
2
FPG Child Development Institute at UNC-Chapel Hill
RTI International
17. Parents of Children with Autism Spectrum
Disorders Have Merited Concerns About Their
Infant Siblings
Caitlin R. McMahon
Elizabeth E. Malesa
Paul J. Yoder
Wendy L. Stone
Vanderbilt University, Psychology and Human Development,
& Vanderbilt Kennedy Center
Poster Session 1 continued on next page
12
2007 Gatlinburg Conference
W E D N E S D A Y
March 7, 2007
18. Personality, Coping Style, and Adjustment of
Parents Rearing Children with Developmental
Disabilities
Angela L. Natcher
Brian M. Jobe
Laraine M. Glidden
24. The Effects of Prematurity on Concept
Formation at Five Years of Age
Vicki Sudhalter
Richard C. Belser
Judith M. Gardner
Bernard Z. Karmel
Department of Psychology, St. Mary’s College of Maryland
N.Y.S. Institute for Basic Research in Developmental
Disabilities
19. The Effect of Disability Status and Social
Experience on Social Cognitive Development
Irene Ngai1
Frank J. Floyd1
Janis B. Kupersmidt2
1
2
Georgia State University
University of North Carolina at Chapel Hill
20. Emotional and Behavioural Adjustment in
Siblings of Children with Intellectual Disability With
and Without Autism
Michael A. Petalas
Susie Nash
Tracey Lloyd
School of Psychology, University of Wales, Bangor
21. Health Knowledge and Expected Outcomes of
Risky Behaviour: A Comparative Study of NonDisabled Adolescents and Young People with
Intellectual and Physical Disabilities
Pownall, J
Jahoda, A
Wilson
25. Interactive Training on Active Support:
Perspectives from Staff, Managers, and Trainers
Vaso Totsika
Sandy Toogood
Susie Nash
University of Wales, Bangor
26. Life Events and Psychological Outcomes in
Adults with Intellectual Disabilities: Longitudinal
Relationships
Lee Williams
Richard Hastings
Stephen Noone
Lauren Burns
Jessica Day
John Mulligan
School of Psychology, University of Wales Bangor
University of Glasgow, Scotland, UK
Section of Psychological Medicine, Division of Community
Based Sciences, Gartnavel Royal Hospital
22. Parental Coping Assistance with Peer Problem
Solving for Children with Intellectual Disabilities
Kara Snead1
Frank J. Floyd1
Janis Kupersmidt2
1
2
Georgia State University
University of North Carolina at Chapel Hill
23. Is Maternal Responsivity Different in Mothers of
Young Children with Fragile X Syndrome Compared
to Down Syndrome?
Audra Sterling
Nancy Brady
Steven F. Warren
University of Kansas
13
2007 Gatlinburg Conference
T H U R S D A Y
March 8, 2007
8:45 A.M. – 10:15 A.M.
• SYMPOSIUM 7 – Ballroom A
Selective Neuropsychological Changes Across the
Adult Lifespan in Down Syndrome
Chair: Darlynne A. Devenny, New York State Institute for
Basic Research in Developmental Disabilities
Discussant: A.J. Holland,University of Cambridge
Autism: Predictors of Response to Treatment as
Behavioral Phenotypes
Travis Thompson1
Glen Sallows2
1
Department of Pediatrics Autism Program, University of
Minnesota School of Medicine
2
Wisconsin Early Autism Project
Declines in Visuo-spatial Abilities in Young Adults
with Down Syndrome
D.A. Devenny
P. Kittler
S.J. Krinsky-McHale
New York State Institute for Basic Research in
Developmental Disabilities
Behavioral Phenotype in Relationship to Analysis of
Behavioral Function and Intervention
William J. McIlvane
Curtis K. Deutsch
Krista M. Wilkinson
William V. Dube
Visuo-spatial Working Memory Decline Associated
with MCI and DAT
S.J. Krinsky-McHale1
P. Kittler1
W. Silverman2
D.A. Devenny1
• SYMPOSIUM 9 – Ballroom C
1
New York State Institute for Basic Research in
Developmental Disabilities
2
Kennedy Krieger Institute and John Hopkins University
School of Medicine
Neuropathological Evidence of Alzheimer’s Disease
in Down Syndrome
L.D. Nelson
V. Kepe
P. Siddarth
J. Barrio
G. Small
University of Massachusetts Medical School - Shriver Center
Biobehavioral Analyses of Self-Injurious Behavior
Chair: Frank J. Symons, Department of Educational
Psychology, University of Minnesota
A Comparison of the Frequency, Location, and
Function of Self-Injurious Behavior between High
and Low Intensity SIB Cases
Chin-Chih Chen
University of Minnesota
A Comparison of the Frequency of Nonverbal
Behavioral Indices of Pain Expression between SIB
and Control Cases During Sensory Testing
Stacy Danov
University of Minnesota
University of California Los Angeles
Integrating Autonomic Arousal into Functional
Behavioral Analysis of Self-Injury
John Hoch
• SYMPOSIUM 8 – Ballroom B
University of Minnesota
Behavioral Phenotypes
Chair: Travis Thompson, Dept of Pediatrics Autism
Program, University of Minnesota School of Medicine
A Comparison of Epidermal Morphology and
Physiology between SIB and Control Cases
Frank Symons
Prader-Willi Syndrome, Oxytocin, and Behavior.
Elisabeth Dykens
Elizabeth Roof
Elizabeth Pantino
Rebecca Kossler
Ray Johnson
Mayim Bialik
Hossein Nazarloo
Sue Carter
University of Minnesota
Vanderbilt Kennedy Center
Ira Lott, MD
10:30 A.M. – 12:00 P.M.
• PLENARY SESSION–REGATTA BALLROOM
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
"Down Syndrome and Alzheimer’s
Disease: Clues from Neuroimaging"
Associate dean of clinical neurosciences, director of the
Division of Child Neurology, and professor of pediatrics and
neurology at the University of California, Irvine
14
2007 Gatlinburg Conference
T H U R S D A Y
March 8, 2007
1:15 P.M. – 2:45 P.M.
• PLENARY SESSION–REGATTA BALLROOM
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
"Social Cognition In Children with Down
Syndrome: Current Evidence,
Future Directions"
Jennifer G. Wishart, PhD
Professor of developmental disabilities in childhood at the
Moray House School of Education, University of Edinburgh
3:00 P.M.– 5:00 P.M.
• PAPER SESSION 1 – Ballroom A
FRAGILE X AND AUTISM
Educational & Therapeutic Involvement by Parents
of Young Children with ASD
Paul R. Benson
Center for Social Development & Education, University of
Massachusetts
Neural Network Analysis of MAOA, Serotonin
Transporter, and Serotonin Receptor (HTR1B)
Polymorphisms on the Behaviors of Children on the
Autism Spectrum
Cohen, I.L.1
Liu, X.2
Jenkins, E.C.1
Brown, W.T. 1
Gonzalez, M. 1
Rovito-Gomez, T. 1
Kim, S-Y1
Lewis, S. 2
Chudley, A. 2
ASD-CARC and Holden, J.J.A.2
1
NYS Institute for Basic Research in Developmental
Disabilities
2
Queens University, Kingston, Ontario
Insomnia in Children with Autism Spectrum
Disorders: Evidence from Multiple Sources
Lynnette M. Henderson
Beth A. Malow
Mary L. Marzec
Susan G. McGrew
Lily Wang
Wendy L. Stone
Girls With Fragile X Syndrome: Early Development
and Behavior
Deborah D. Hatton1
John Sideris1
Anne Wheeler1
Kelly Sullivan2
1
2
The University of North Carolina at Chapel Hill
Duke University
Negative Reactivity in Preschool Boys with Fragile
X Syndrome
Jane E. Roberts
Meghan Shanahan
Deborah D. Hatton
University of North Carolina at Chapel Hill
Psychotropic Medication Use in Fragile X
Syndrome: Analysis of Side Effects
Maria G. Valdovinos
Rahul A. Parsa
Michelle L. Alexander
Drake University
• PAPER SESSION 2 – Ballroom B
FAMILIES, MENTAL HEALTH, AND
SOCIAL BEHAVIOR
The Pivotal Behavior Model of Development:
Implications for Developmental Intervention
Gerald Mahoney
Frida Perales
Mandel School of Applied Social Sciences, Case Western
Reserve University
Predicting Disruptive Behavior Disorders in FiveYear Old Children with and without Intellectual
Disability
Bruce L. Baker1
Rachel Fenning1
Jan Blacher2
1
Department of Psychology, UCLA
Graduate School of Education, University of California,
Riverside
2
Mental Health of Children and Adolescents with and
without Intellectual Disabilities in Britain
Eric Emerson
Chris Hatton
Institute for Health Research, Lancaster University, UK
Vanderbilt Kennedy Center for Research in Human
Development
Paper Session 2 continued on next page
15
2007 Gatlinburg Conference
T H U R S D A Y
March 8, 2007
Household Task Participation of Children with and
without Attentional Problems
M. Louise Dunn1
Wendy J. Coster2
Gael S. Orsmond2
Ellen S. Cohn2
1
2
University of Utah
Boston University
Using Electropalatograpy (EPG) to Assess and Treat
Speech Articulation Difficulties in Children with
Down’s Syndrome
Sara Wood1
Bill Hardcastle1
Joanne McCann1
Claire Timmins1
Jennifer Wishart2
1
Socio-Economic Position, Social Connectedness
and the Self-Reported Well-Being of Women and
Men with Intellectual Disabilities in England
Chris Hatton
Eric Emerson
Institute for Health Research, Lancaster University, UK
Unmet Need in Families of Young People with
Intellectual and Developmental Disabilities
Vivien J Moffat 1
Jennifer G Wishart 2
Eve C Johnstone 1
1
2
Division of Psychiatry, University of Edinburgh
Moray House School of Education, University of Edinburgh
• PAPER SESSION 3 – Ballroom B
DOWN SYNDROME AND
INTERVENTIONS
Telomere Shortening in Chromosomes 21, 1, 2, and
16, and Reduced Signal Number in T-Lymphocyte
Metaphases from Older Individuals with Down
Syndrome and Dementia
E.C. Jenkins1
M.T. Velinov1
L. Ye1
H. Gu1
Samantha A. Ni1
D. Pang1,2
D.A. Devenny1
W.B. Zigman1
N. Schupf1,2
W.P. Silverman1,3
1
NYS Institute for Basic Research in Developmental
Disabilities
2
Taub Institute for Research on Alzheimer’s Disease and the
Aging Brain, Columbia University
3
Kennedy-Krieger Institute, Johns Hopkins University School
of Medicine
Down’s Syndrome, Ageing and Alzheimer’s Disease
Holland, A.J.
Ball, S.
Herbert, J.
University of Cambridge, UK
16
Queen Margaret University College, Edinburgh
University of Edinburgh
2
Inclusive Education and Social Isolation among
Adolescents with Down Syndrome
Joanne Kersh
Penny Hauser-Cram
Helena Pan
Boston College
Establishing the Building Blocks of Cognitive
Behavioural Therapy with People with Mild
Learning Disabilities: Developing a Collaborative
Relationship
Andrew Jahoda1
Carol Pert1
Peter Trower2
Biza Stenfert-Kroese2
Dave Dagnan3
Mhairi Selkirk1
1
University of Glasgow
University of Birmingham
3
University of Lancaster
2
5:15 P.M. – 6:15 P.M.
• PLENARY SESSION–REGATTA BALLROOM
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
12345678901234567890123456789012123456789012345678901234567890121234567890
UPDATE ON NIH TASK FORCE ON
DOWN SYNDROME
2007 Gatlinburg Conference
T H U R S D A Y
March 8, 2007
Angeles
6:30 P.M. – 8:30 P.M.
• POSTER SESSION 2– ATRIUM
1. Language Development in Toddlers with
Developmental Disabilities Following Intervention:
The Role of Child and Parent Communication
Characteristics
R. Micheal Barker
Rose A. Sevcik
Georgia State University
7. Stressful Social Interactions, Attributions, and
Coping among Depressed Adults with Mild
Intellectual Disability-Poster Presentation
Sigan L. Hartley
William E. MacLean, Jr.
University of Wyoming
8. Family Cohesion and Its Influence on Adaptive
Behavior Development in Children with a Disability
Lara S. Head1
Alan M. Gross2
1
2. Repetitive Behaviors and Sensory Features in
Young Children with Autism: Examining
Relationships and Specificity
Brian A. Boyd1
Grace T. Baranek
Linda R. Watson
Elena Gay
University of North Carolina at Chapel Hill
2
Waisman Center, University of Wisconsin-Madison
The University of Mississippi
9. Father Presence as a Predictor of Maternal Stress
and Quality of Life in Families with Children with
Fragile X Syndrome
Anna James
Jane E. Roberts
Don Bailey
University of North Carolina at Chapel Hill
3. Predicting the Depression and Well-being of
Mothers Rearing Children with Developmental
Disabilities
April M. Corrice
Laraine M. Glidden
Brian M. Jobe
St. Mary’s College of Maryland
4. Positive States as Mediators of Stress in Parents
of Children with Different Types of Intellectual
Disabilities
Elizabeth Roof
Elizabeth Pantino
Rebecca Kossler
Ray Johnson
Elisabeth M. Dykens
Vanderbilt Kennedy Center
10. Auditory Brainstem Evoked Responses in
Newborns with Down Syndrome
P. Kittler1, 2
B.Z. Karmel1, 2
H.T.T. Phan1, 2
J.M. Gardner1, 2
A. Gordon1, 2
A. Harin2
1
2
NYS Institute for Basic Research in DD
St. Vincent’s Catholic Medical Center
11. Gender and Delay of Gratification in High Risk
Children
Shira Kolnik
Ann-Marie Faria
Marygrace Yale Kaiser
University of Miami
5. Insecure and Disorganized Attachment among AtRisk Children: The Influence of Subthreshold
Neglect
Jaelyn R. Farris
Jody S. Nicholson
John G. Borkowski
University of Notre Dame
12. Changes in Joint Attention Following Two Years
of Early Intensive Behavioural Intervention in
Preschool Aged Children with Autism
Hanna Kovshoff1
Richard Hastings2
Bob Remington1
1
6. The Role of Caregiver Praise as a Mechanism for
Children’s Sustained Engagement
Kim Har
Connie Kasari
2
School of Psychology, University of Southampton, UK
School of Psychology, University of Wales, Bangor, UK
Graduate School of Education, University of California, Los
Poster Session 2 continued on next page
17
2007 Gatlinburg Conference
T H U R S D A Y
March 8, 2007
13. Maternal Predictors of Adaptive Behavior of
Boys with Fragile X Syndrome
Heather Magruder
FPG Child Development Institute, University of North
Carolina at Chapel Hill
14. Autism in Early Childhood: Identification and
Treatment
Nicole J. Neudorfder
Aila Dommestrup
Laura Lee McIntyre
Syracuse University
15. Trajectory of Mathematics and Reading Skills in
School-age Girls with Fragile X Syndrome
Melissa M. Murphy
Michèle M. M. Mazzocco
Johns Hopkins University and Kennedy Krieger Institute
16. Lip Dynamics and Stereotypic Movement
Disorders
Karl M. Newell1
F. Aileen Costigan1
James W. Bodfish2
1
2
The Pennsylvania State University
University of North Carolina, Chapel-Hill
17. A Longitudinal Examination of the Mediating
Role of Behavior Problems in Adaptation for
Families of Children With and Without
Developmental Delays
Anita Pedersen y Arbona
Keith Crnic
Arizona State University
18. Cross-Syndrome Comparisons of Gesture Use in
Toddlers
Amy Philofsky1
Susan L. Hepburn1
Deborah J. Fidler2
Sally J. Rogers3
1
Department of Psychiatry, University of Colorado at Denver
Health Science Center
2
Human Development & Family Studies, Colorado State
University
3
MIND Institute, Department of Psychiatry, University of
California, Davis
19. Down Syndrome, and Congenital Heart Defect:
Maternal Experiences of Bereavement
Deirdre Reilly
Frances Vaughan
Jaci Huws
School of Psychology, University of Wales, Bangor
18
20. Correlations between Locus of Control and
Assertiveness and Knowledge of Abuse and
Decision-Making Performance among Individuals
with Moderate to Severe Disabilities
Anne Spillane
National University
21. Sibling Relationship Quality for Individuals with
Mental Retardation: A Comparison of Sibling
Reports
Shana Strickland
Vivian Piazza
Frank J. Floyd
Georgia State University
22. Examining Contingency Control Deficits in
Children with and without Autism
Louis P. Hagopian1
Lisa M. Toole2,3
Eric Boelter2
Heather K. Jennett2
Gregory A. Lieving4
Keith J. MacWhorter2
Elizabeth L. Tobin5
1
The Johns Hopkins University School of Medicine
Kennedy Krieger Institute
3
University of Maryland, Baltimore County
4
West Virginia University Institute of Technology
5
Towson University
2
23. Understanding of the Causes of Emotions in
Others by Children with Down’s Syndrome
K.R. Williams1
J.G. Wishart1
D.S. Willis2
T.K. Pitcairn1
1
2
University of Edinburgh
Napier University
24. Imitation in Young Children with Autism and
Other Developmental Delays
Connie S. Wong
Cleveland State University
25. The Relationship Among Vocabulary Knowledge,
Phonological Awareness, and Reading Performance
in Children with Mild Intellectual Disabilities
Rose A. Sevcik
Justin C. Wise
Robin D. Morris
Georgia State University
2007 Gatlinburg Conference
F R I D A Y
March 9, 2007
8:45 A.M. – 10:15 A.M.
• SYMPOSIUM 10 – Ballroom A
Individual Differences in Adaptation in Parents and
Siblings of Children with ID – Process Mechanisms
and Implications for Support
Chair: Malin B Olsson, Department of Psychology,
Goteborg University
Longitudinal Perspectives on Sibling Impact: The
Role of Clinical Behavior Problems
Jan Blacher1
Cameron Neece2
1
2
University of California, Riverside
UCLA
Acceptance as a Longitudinal Predictor of Stress,
Anxiety and Depression in Mothers of Children with
Intellectual Disabilities
Tracey Lloyd
Richard Hastings
University of Wales, Bangor
Family Process in Families of Young Children with
Intellectual Disabilities: Depression and InterParental Conflict
Zolinda Stoneman
Institute on Human Development and Disability/
UCEDD,University of Georgia
The Impact of Psychological Variables on WellBeing of Parents of Children with ID
Malin B Olsson
Department of Psychology,Goteborg University
• SYMPOSIUM 11 – Ballroom B
Phenotypic Responses to Communication
Intervention by Young Children with Down
Syndrome
Chair: Steven F. Warren, Institute for Life Span Studies,
University of Kansas
Discussant: Len Abbeduto, Waisman Center, University
of Wisconsin at Madison
Response by Children with Down Syndrome to
Prelinguistic Milieu Teaching
Nancy C. Brady1
Steven F. Warren1
Marc E. Fey2
Lizbeth H. Finestack2
Shelley L. Bredin-Oja2
Kandace K. Fleming1
1
Institute for Life Span Studies, University of Kansas,
Lawrence, KS
2
University of Kansas Medical Center, Kansas City, KS
Effects of Early Augmented Language Intervention
on the Communication Skills of Young Children
with and without Down Syndrome
Mary Ann Romski
Rose A. Sevcik
Lauren B. Adamson
Melissa Cheslock
Ashlyn Smith
Mike Barker
Roger Bakeman
Georgia State University
• SYMPOSIUM 12 – Ballroom C
What Large-Scale, Administrative Databases
Can Tell Us about Down Syndrome
Chair: Robert M. Hodapp, Vanderbilt Kennedy Center for
Research on Human Development, Department of Special
Education, Peabody College, Vanderbilt University
Using State Administrative Databases to Study
Down syndrome
Richard C. Urbano
Vanderbilt Kennedy Center for Research on Human
Development, Department of Pediatrics, Vanderbilt
University Medical Center
The Demography of Births in Down Syndrome
Robert M. Hodapp
Vanderbilt Kennedy Center for Research on Human
Development, Department of Special Education, Peabody
College, Vanderbilt University
Treatment (non)Effects and Predictors of
Communication and Language Development in
Children with Down Syndrome
Paul J. Yoder1
Steven F. Warren2
Down Syndrome and Adverse Birth Outcomes
S. Trent Rosenbloom
1
Propensity Scores: A Method for Matching on
Multiple Variables in Down Syndrome
Jennifer U. Blackford
John F. Kennedy Center for Human Development,
Vanderbilt University
2
Institute for Life Span Studies, University of Kansas
Vanderbilt Kennedy Center for Research on Human
Development, Department of Biomedical Informatics
Vanderbilt Kennedy Center for Research on Human
Development, Department of Psychiatry, Vanderbilt
University Medical Center
19
2007 Gatlinburg Conference
F R I D A Y
March 9, 2007
10:30 A.M. – 12:00 P.M.
• PLENARY SESSION–REGATTA BALLROOM
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
123456789012345678901234567890121234567890123456789012345678901212345678901
"Therapy for Structural Anomalies in the
Trisomic Brain"
Roger H. Reeves, PhD
Professor in the Department of Physiology at the Johns
Hopkins University School of Medicine and Institute for
Genetic Medicine
12:00 P.M.
• PLENARY SESSION–REGATTA BALLROOM
CLOSING REMARKS
Marsha Mailick Seltzer, PhD & Len Abbeduto, PhD
Waisman Center, University of Wisconsin-Madison
20
Symposium 1
S Y M P O S I U M
1
Early Risk for Developmental Delays among Children of First
Time Mothers: Two Year Findings from the Parenting for the
First Time Project
Chair:
John G. Borkowski
University of Notre Dame
Discussant: Craig Ramey
Georgetown University
21
SYMPOSIUM 1
Early Risk for Developmental Delays among Children of First Time Mothers:
Two Year Findings from the Parenting for the First Time Project
Chair:
John G. Borkowski, University of Notre Dame
Discussant: Craig Ramey, Georgetown University
The Parenting for the First Time Project: Design and Methodology
Jennifer Burke Lefever1,Robin Gaines Lanzi2,Centers for the Prevention of Child Neglect3
1
University of Notre Dame,2 Georgetown University,3 Georgetown University, University of
Alabama-Birmingham, University of Kansas, University of Notre Dame, University of Texas at
Houston
Prenatal Cumulative Risk and Maternal Parenting During Infancy
Kimberly S. Howard1,Judith J. Carta2,Kere P. Hughes3,Julie N. Schatz4,John G.
Borkowski4,Centers for the Prevention of Child Neglect5
1
Teachers College, Columbia University,2University of Kansas,3Iowa State University,4University of
Notre Dame,5Georgetown University, University of Alabama-Birmingham, University of Kansas,
University of Notre Dame, University of Texas at Houston
Application of the E-RISK Index with Children with Special Needs: A Reliable and Easy-to
Administer Screening Tool of Early Precursors of Likely Neglectful Behaviors
Robin Gaines Lanzi1,Sharon Landesman Ramey1,Shannon Carothers1,Bette R. Keltner1,Centers
for the Prevention of Neglect2
1
Georgetown University,2Georgetown University, University of Alabama-Birmingham, University of
Kansas, University of Notre Dame, University of Texas at Houston
The Longitudinal Impact of Maternal Warmth and Hostility on Early Language Development:
Comparisons of Adolescent and Adult Mothers
Leann E. Smith1,Gitta Lubke2,Steve Warren3,Centers for the Prevention of Child Neglect4
1
University of Wisconsin-Madison,2University of Notre Dame,3University of Kansas,4Georgetown
University, University of Alabama-Birmingham, University of Kansas, University of Notre Dame,
University of Texas at Houston
Prenatal Maltreatment Risk, Early Parenting Behaviors, and Children’s Emergent
Regulation
Julie Schatz1,John G. Borkowski1,Centers for the Prevention of Child Neglect2
1
University of Notre Dame,2Georgetown University, University of Alabama-Birmingham, University
of Kansas, University of Notre Dame, University of Texas at Houston
Obstacles to Service Receipt for Children with Early Indicators of Developmental Delays
Carol E. Akai1,Leann E. Smith2,Lorraine V. Klerman3, Bette R. Keltner4, Kristi C. Guest5,Centers
for the Prevention of Child Neglect6
1
University of Notre Dame,2University of Wisconsin—Madison,3Brandeis University,4Georgetown
University,5University of Alabama at Birmingham,6Georgetown University, University of AlabamaBirmingham, University of Kansas, University of Notre Dame, University of Texas at Houston
22
SYMPOSIUM 1
The Parenting for the First Time Project: Design and Methodology
Jennifer Burke Lefever1, Robin Gaines Lanzi2, and the Centers for the Prevention of Neglect3
1
University of Notre Dame; 2Georgetown University; 3Georgetown University, University of
Alabama-Birmingham, University of Kansas, University of Notre Dame, University of Texas at
Houston
Contact: Jennifer Burke Lefever, 940 Flanner, Notre Dame, IN 46556, [email protected]
Introduction: The Parenting for the First Time Project was designed to assess the impact of varying
degrees and types of neglect and poor parenting on children’s development during the first 3 years of life,
including changes in intelligence and behavior, language, social and emotional well-being, physical
growth, and health status. Data from this study will also help to test and further refine the Notre Dame
Adolescent Parenting Conceptual Model by expanding the diversity of teen parents and community
settings studied, by including a broad array of assessments related to the construct of childhood neglect,
and by applying sophisticated analytic strategies to test the developmental associations among parenting
characteristics, parenting behaviors and attitudes, and child developmental delays in multiple domains.
Methods: Six hundred and eighty-two expectant mothers were recruited during pregnancy through
primary care facilities in the communities of Birmingham, AL, Kansas City, KS, South Bend, IN, and
Washington, D.C. Three different groups of first-time mothers were included in the sample: adolescents
(n=396), low-ed adults (less than 2 years formal education beyond high school; n=169), and hi-ed adults
(at least 2 years of formal education; n=117). The age at the time of the first child’s birth ranged from
14.68 to 36.28, with an average of 17.49 for the adolescents, 25.48 for the low-ed adults, and 27.88 for
the hi-ed adults. Approximately 65% of the sample is African-American, 19% is White/Non-Hispanic, 15%
is Hispanic, 1% is multi-racial, and .5% is of an other race. The adolescent and low-ed adult samples are
closely matched on race/ethnicity.
Mothers were interviewed in their last trimester of pregnancy as well as when their children were 4, 6, 8,
12, 18, 24, 30, and 36-months old. Interviews at the prenatal, 6, 12, 24, and 36-month visits primarily
focused on risks for poor parenting, such as maternal depression (Beck II), parenting stress (Parenting
Stress Index – Short Form), and lack of social support; parenting beliefs and practices; as well as other
demographic information. The 4, 8, 18, and 30-month visits occurred in the home and included both
interviews and observations of parenting practices (Home Observation for Measurement of the
Environment, Supplement to the HOME for Impoverished Families, Landry Naturalistic Observation, and
Observation of Parenting Essentials). After each of the home visits, mothers were given a cellular phone
and interviewed multiple times over the phone concerning their daily parenting practices (Parent-Child
Activities Interview). At the 12, 24, and 36-months visits, the children were also tested for intellectual
(Bayley II) and language abilities (Pre-School Language Scales – IV), rated on their behavior by both their
mother (Infant Toddler Social and Emotional Assessment) and child tester (Bayely Behavioral Rating Scale
II), and their height and weight were measured. Upon completing each assessment after the child’s birth,
the interviewers also rated the child’s environment for risks of physical neglect.
Discussion: This study represents one of the first-ever prospective broad-based, multi-site investigations
of child neglect among a diverse sample of adolescent mothers and will help to establish a foundation for
future preventive interventions to reduce the incidence and impact of neglect and abuse on child
development. This data set identifies risks for developmental delay as well as protective factors,
measured in detail, to better map the multiple and fluctuating social ecologies and personal life
circumstances of teen mothers and their young children. Results from this project can be used to identify
the mechanisms leading to environmental risks for early developmental disabilities.
23
SYMPOSIUM 1
Prenatal Cumulative Risk and Maternal Parenting During Infancy
Kimberly S. Howard1, Judith J. Carta2, Kere P. Hughes3, Julie N. Schatz4, John G. Borkowski4, and the
Centers for the Prevention of Child Neglect51Teachers College, Columbia University; 2University of Kansas;
3
Iowa State University; 4University of Notre Dame, 5Georgetown University, University of AlabamaBirmingham, University of Kansas, University of Notre Dame, University of Texas at Houston
Contact: Jennifer Burke Lefever, 940 Flanner, Notre Dame, IN 46556, [email protected]
Introduction: Much research over the past twenty years has substantiated the fact that children’s
development is affected by a number of individual and environmental factors. Theories of cumulative risk
posit that individual risks may not greatly influence the course of child development, but when risk factors
accumulate, their aggregate effect adversely impact a child’s developmental trajectory (e.g., Rutter,
1979). Few studies have examined the relationship between cumulative risk and parenting outcomes.
Recently, Nair and colleagues (2003) studied cumulative risk in a sample of substance abusing women
and found significantly higher levels of parenting stress and child abuse potential for women with five risks
compared to those with four or fewer risks. Similarly, the present study sought to examine how cumulative
risk assessed during pregnancy related to parenting outcomes assessed during the earliest stages of their
parenting experience—when their children were 4 and 8 months of age.
Methods: Participants included 682 mother/infant dyads who were part of a larger longitudinal study of
first time parenting. The sample was divided into two subsamples: Those with less than 2 years of college
education (n=565) and those with at least 2 years of college (n=117). The primary analyses were
conducted using only the low-education sample in order to identify the effects of risk in an already at-risk
group. Maternal risk status was assessed prenatally in six domains: education and intelligence, maternal
cognitive readiness to parent, maternal socio-emotional functioning, health and stress, maltreatment risk
and demographic risks. The cumulative risk index consisted of a sum of 22 dichotomous risks.
Results: Overall, it was found that low-education mothers (M=5.07; SD=2.8) experienced more risks than
higher-education mothers (M=2.03; SD=2.0), but that even among low-education mothers, the quantity
and type of risks that they experienced prenatally were related to maternal parenting at both 4, β = -.228,
p < .01, R2 = .052, and 8 months, β = -.350, p < .01, R2 = .122. Mothers who experienced fewer risks on
the index were rated as being more positive and less negative with their infants. They also provided higher
quality home environments and more regularly engaged in parenting “essentials.” Subscale analyses
revealed that the maternal readiness subscale was the most significant in predicting parenting at 4, β = .256, p < .01, and 8 months, β = -.325, p < .01, accounting for 6.5% and 10.5% of the variance,
respectively. Within maternal readiness, child abuse potential and parenting styles were the most
important variables. In combination, these two variables accounted for 9.8% of the variance in the
parenting composite at 4 months and 13% of the variance at 8 months. Finally, total number of risks was
associated with parenting quality such that mothers who reported a greater number of risks were less
likely than mothers with fewer risks to engage in optimal parenting practices with their infants (p < .05).
Discussion: The results suggest that prenatal cumulative risk has implications for the quality of maternal
parenting during infancy, which is likely a mediating force for the relationship between parent sociodemographic risk and children’s developmental outcomes that has been documented in the literature. It is
possible that identifying and intervening with maternal risks early on, particularly those related to cognitive
readiness for parenting, could significantly improve both maternal parenting practices and later
developmental outcomes for children.
24
SYMPOSIUM 1
Application of the E-RISK Index with Children with Special Needs: A Reliable and Easy-to
Administer Screening Tool of Early Precursors of Likely Neglectful Behaviors
Robin Gaines Lanzi1, Sharon Landesman Ramey1, Shannon Carothers1, Bette R. Keltner1, and the
Centers for the Prevention of Neglect2
1
Georgetown University; 2Georgetown University, University of Alabama-Birmingham, University of
Kansas, University of Notre Dame, University of Texas at Houston
Contact: Jennifer Burke Lefever, 940 Flanner, Notre Dame, IN 46556, [email protected]
Introduction: Children who are neglected have been shown to suffer adverse psychological and
educational outcomes (De Bellis, 2005). With approximately 3 million referrals to Child Protective
Services concerning the welfare of approximately 5 million children annually and about 879,000 of them
substantiated cases of maltreatment (National Clearinghouse on Child Abuse and Neglect, 2002), this is a
major issue. Although conceptual and operational definitions of child neglect have been discussed widely
in the literature (e.g., Dubowitz et al. 2005; Runyan et al., 2005), no standardized operational definition of
or method for identifying child neglect exists (Zuravin, 1998). This stems from a number of factors
including varying state guidelines, differing theoretical research perspectives, and lack of sound
methodological tools. Clearly, there is a need to adopt a more practically useful methodology for
measuring both the risk for and the occurrence of child neglect so that practitioners, educators, and
service providers can more reliably identify children at risk for child neglect and provide resources and
services to prevent child neglect and associated poor developmental outcomes.
Methods: The present study addresses this gap by developing a practically useful 4-item risk index of
likely neglectful behaviors that can be administered during the prenatal period. The E-RISK index was
developed as part of the Centers for the Prevention of Neglect — a 4-site prospective study of the early
predictors and precursors of neglect in the first 3 years of life among children born to 682 adolescent and
adult mothers.
Results: The 4-item E-RISK index of early precursors of likely neglectful behaviors was comprised of 4
questions administered during the prenatal interview concerning: recognition and acceptance of new
responsibility as a parent; certainty about ability to provide emotional support to child; certainty about
ability to do good job raising child; and beliefs about parenting (i.e., indicate young children who feel
secure often grow up not expecting too much). Negative responses to these questions were summed for
the index. There was variation by mom group in the number of risk factors: 0 risk - 37% teen, 36% adult
low, 60% adult high; 1 risk – 42% teen, 47% adult low, 35% adult high; 2 risks – 16% teen, 15% adult low,
5 % adult high; 3-4 risks 4% teen, 3% adult low, and 0 % adult high (p<.001). The E-RISK Index was
significantly related to the home environment and independently observed mother-child interactions at the
4, 8, and 18 month home visits (see Table 1). The E-RISK index significantly predicted the child’s
development at 24 months, particularly for the children of teen mothers in terms of their social and
emotional development (see Table 2). We further examined which children were scoring 1 ½ SD below
the mean on the Bayley and PLS and found that the E-RISK was a significant predictor for children of
adult mothers (p<. 01) but not teen mothers.
Discussion: We will discuss how we can reasonably adapt these questions as a screening tool for service
providers and clinicians to best meet children’s developmental needs as well as supportive needs for
children and families.
Key References:
De Bellis, M. D. (2005). The psychobiology of neglect. Child Maltreatment, 10, 150-172.
Dubowitz H et al. (2005). Examination of a conceptual model of child neglect. Child Maltreatment, 10, 173-189.
Zuravin, S. J. (1998). The ecology of child abuse and neglect: Review of the literature and presentation of data.
Violence and Victims., 4(2), 101-120. Table 1. E-RISK Index is Significantly Related to HOME Environment and
Mother-Child Interactions.
25
SYMPOSIUM 1
The Longitudinal Impact of Maternal Warmth and Hostility on Early Language
Development: Comparisons of Adolescent and Adult Mothers
Leann E. Smith1, Gitta Lubke2, Steve Warren3, and the Centers for the Prevention of Child
Neglect University of Wisconsin-Madison; 2University of Notre Dame; 3University of Kansas; 4Georgetown
University, University of Alabama-Birmingham, University of Kansas, University of Notre Dame, University
of Texas at Houston
Contact: [email protected]
Waisman Center, 1500 Highland Ave., Madison, WI 53705
41
Introduction: The present study investigated changes in, and correlates of, maternal warmth and hostility
during the first 18 months of life using latent growth curve (LGC) modeling. Participants included 682
mother-infant dyads from a multi-site, longitudinal study of first time mothers and their children, were
recruited from hospitals, health clinics, social service agencies, and school-aged mothers’ programs. The
sample consisted of 396 adolescent mothers and 286 adult mothers, ranging in age from 15 to 35 years
(mean age = 20.73). Sixty-four percent of mothers were African-American, 18% Caucasian, 15% Latina,
and 3% Multiracial/Multiethnic or Other. Most families were living in poverty with approximately 50% of
mothers reporting annual incomes of $15,000 or less, 25% reporting between $15,000 and $30,000, and
25% reporting over $30,000.
Methods: Mothers reported on expectations of paternal support and cognitive readiness to parent during
the prenatal period. When children were 4, 8, and 18 months of age, mothers were rated on their warmth
and hostility towards children during naturalistic home observations. The Preschool Language Scale, 4th
Edition was used as a measure of children’s expressive communication and auditory comprehension at 24
months. In comparison with national norms (M=100, SD=15), children in both groups demonstrated belowaverage language scores. Children of teens had significantly lower average expressive language scores
(M=91.6, SD=13.2) than children of adults (M=96.4, SD=12.1) and marginally significant lower scores on
auditory language (M=90.3, SD=14.9) than children of adult mothers (M=94.2, SD=15.0), Furthermore,
43% of children of teens scored at least one standard deviation below the standardized mean on auditory
comprehension, in comparison with 29% of adults. Similarly, 23% of children of teens were a standard
deviation below the mean on expressive communication, in contrast with 12% of children of adults.
Results: LGC analyses revealed that for both adolescent and adult groups, warmth did not change from 4
to 18 months, whereas hostility was increasing over the same time period. Adolescents, however, were
becoming hostile more quickly than adults. Adolescents also had lower levels of initial warmth and higher
levels of initial hostility than adults. Rate of change in hostility was a marginally significant predictor of
expressive language for adolescents, but not adults, such that teens who changed at a slower rate had
children with more-optimal language development. Maternal expectations of support from the children’s
fathers were associated with lower levels of hostility at 4 months. High expectations of father support were
also linked with slower increases in hostility over time. Although warmth did not change over time, prenatal
cognitive readiness in both teens and adults was related to higher levels of warmth at 4 months, which in
turn, predicted children’s expressive language at 24 months.
Discussion: The present study highlights how different dimensions of parenting change uniquely across
time; in this case, maternal warmth was stable from 4 to 18 months, whereas hostility increased as
children aged. The current findings also suggest that although adolescents may display lower levels of
positive parenting behaviors than adults, the parenting processes appear to be similar across groups. As
such, new mothers, regardless of their age, may benefit from interventions aiming to increase cognitive
readiness and social support.
26
SYMPOSIUM 1
Prenatal Maltreatment Risk, Early Parenting Behaviors, and Children’s Emergent Regulation
Julie Schatz1, John G. Borkowski1, and the Centers for the Prevention of Child Neglect21University of Notre
Dame; 2Georgetown University, University of Alabama-Birmingham, University of Kansas, University of
Notre Dame, University of Texas at Houston
Contact: [email protected], 1602 N. Ironwood Dr., South Bend, IN 46635
Introduction: There is consistent research evidence supporting parenting behaviors as the mechanisms
responsible for compromising children’s subsequent regulation. Because the effects of maltreatment on
children’s regulation is well-documented in school-aged children with evidence suggesting even earlier
dysfunction, elucidating the relationships among maltreatment risk, parenting practices, and toddlers’
developing regulation becomes especially important for the prevention of later childhood psychopathology.
The present study explores the relationship between early maltreatment risk and parenting behaviors over
the course of the first 18 months of life. This study also examines predictors of change in the parenting
behaviors over time and how change serves as the mechanism responsible for early maternal
maltreatment risk’s influence on children’s developing self-regulation.
Methods: One hundred eighty-nine mother-child dyads were drawn from the ongoing Parenting for the
First Time Project which was designed to understand low-levels of neglectful parenting among mothers
and their first-born children. Both laboratory-based and home-based interviews and observations were
employed in the design with assessments completed prenatally, at 4, 6, 8, and 18 months, and 24 months.
During the prenatal visit, maternal child abuse potential was measured with an abbreviated form of the
Child Abuse Potential Inventory (CAPI) and expectations about children’s development and behaviors
were assessed with the Parenting Style Expectations Questionnaire (PSE). Additional maltreatment risk
factors were assessed at 6 months with self-report measures of the Childhood Trauma Questionnaire
(CTQ) and History of Neglect which measures maternal childhood experiences of neglect and abuse. At 4,
8, and 18 months, parenting was assessed by observational ratings of 7 parenting skills (i.e. protect,
comfort, guide, celebrate, language stimulation, mentor/rehearse, recognize/respond, and encourage
exploration). Parenting behaviors, including display of positive affect, warmth/sensitivity, contingent
responsiveness, and general verbalness, were also coded during an observational period. In addition, the
HOME and the Supplemental to the Home Scale for Impoverished Families (SHIF) rating scales were
obtained as measures of the parenting environment. Children’s regulation was measured at 24 months
with both a mother report and an observational rating completed by a child assessor after developmental
testing. The Infant-Toddler Social and Emotional Assessment (ITSEA) measured children’s early emotional
and behavioral regulation abilities. The Bayley Rating Scale (BRS) was also used to index children’s
regulation with items drawn from two factor scales: the emotion regulation and orientation/engagement
scales that address emotion and cognitive regulation components.
Results: The full structural equation model consisted of the constructs of maltreatment risk, parenting at
4, 8, and 18 months, and children’s regulation at 24 months. Early maltreatment risk was expected to
predict early parenting which, in turn, would predict later parenting and later parenting was expected to
predict regulation. Preliminary analyses showed early maltreatment risk to be a significant predictor of
parenting at 4 months. Parenting at 8 and 18 months were each strongly predicted by the prior parenting
time-point. Preliminary results also suggested that certain parenting behaviors changed during the first 18
months of parenting.
Discussion: Overall, the results highlight the role of early maltreatment risk in determining early parenting
which predicts subsequent parenting behaviors and later children’s regulation. Changes in parenting
behaviors were also evident. The results highlight the importance of early risk assessment to optimize
parenting and foster children’s regulatory development.
27
SYMPOSIUM 1
Obstacles to Service Receipt for Children with Early Indicators of
Developmental Delays
Carol E. Akai1, Leann E. Smith2, Lorraine V. Klerman3, Bette R. Keltner4, Kristi C. Guest5, and the
Centers for the Prevention of Child Neglect6
1
University of Notre Dame; 2University of Wisconsin; 3Brandeis University; 4Georgetown University;
5
University of Alabama-Birmingham; 6Georgetown University, University of Alabama-Birmingham,
University of Kansas, University of Notre Dame, & University of Texas at Houston
Contact: [email protected], Notre Dame Center for Children and Families
1602 N. Ironwood Dr., South Bend, IN 46635
Introduction: According to many state guidelines provided by the Individuals with Disabilities Education
Improvement Act of 2004 (PL 108-446), children with at least a 25% delay in cognitive, language, motor,
adaptive, or social-emotional development are eligible for early intervention services. Children also qualify
for services if they have a 20% delay in at least two of these domains. The present study revealed that
although many children qualified for services, few mothers were aware of their children’s developmental
problems, and hardly any children were identified by medical professionals as having problems that may
interfere with development.
Methods: Data were drawn from the multi-site, longitudinal Parenting for the First Time study following
primiparous mothers and their children over time. Teen and adult mothers of infants in South Bend, IN,
Washington, D. C., Birmingham, AL, and Kansas City, KS participated in the study. Children were
ethnically diverse and had high levels of demographic risk for developmental delays; two-thirds of families
reported annual incomes below $20,000 and three-fifths of mothers had not completed high school.
Cognitive development (Bayley Mental), language development (Preschool Language Scale), adaptive
development (Bayley Behavior Ratings), and social-emotional development (Brief Infant-Toddler Social
and Emotional Assessment) were measured for children at 12 months and 24 months of age. During these
times, mothers were interviewed about children’s development as well as indications of developmental
problems from medical professionals.
Results: Based on national norms, children’s delays in each developmental domain were calculated.
According to IDEA guidelines, 13% of children were eligible for intervention services at 12 months and
28% were eligible at 24 months. Of the 49 eligible children at 12 months, only 2 were identified by medical
professionals as having problems that could interfere with development. Likewise, only 9 out of 85 eligible
children at 24 months were identified having problems. Most eligible children were, however, regularly
receiving medical services. Much like doctors, few mothers noticed problems early; at 12 months, only 9%
of mothers reported being “worried” or “very worried” about their children’s behavior, emotions, and
relationships and only 7% reported being “worried” or “very worried” about language development.
Discussion: Percentages of change in delay status within each developmental domain from 12 to 24
months as well as change in overall eligibility status from 12 to 24 months were also determined. More
than one in five children who were not eligible for services at 12 months became eligible at 24 months
(indicating they had “gotten worse”). Yet, for the most part, developmental problems in this at-risk sample
remained unnoticed and untreated. In order to promote optimal development among children in poverty,
families need to be aware of and have access to early intervention services.
28
Symposium 2
S Y M P O S I U M
2
Down Syndrome, Aging, and Alzheimer’s Disease:
Practical Insights from Prospective Studies
Chair:
Wayne Silverman
Kennedy Krieger Institute and Johns Hopkins
University School of Medicine
29
SYMPOSIUM 2
Down Syndrome, Aging, and Alzheimer’s Disease:
Practical Insights from Prospective Studies
Chair:
Wayne Silverman, Kennedy Krieger Institute and
Johns Hopkins University School of Medicine
β42,Incident Dementia and Mortality in Down
Elevated Plasma Amyloid β-Peptide Aβ
Syndrome
Nicole Schupf1, Bindu P. Patel2, Deborah Pang2, Warren B. Zigman3, Pankaj D. Mehta3, Richard P.
Mayeux2, Wayne Silverman4
1
Columbia University, G. H. Sergievsk Center,2Columbia University Medical Center, 3NYS Institute
for Research on Developmental Disabilities,4Kennedy Krieger Institute and Johns Hopkins School
of Medicine
Apolipoprotein ε4 is Associated with Increased Mortality Risk in Nondemented Adults with
Down Syndrome
Warren B. Zigman1,Edmund C. Jenkins1,Benjamin Tycko2,Nicole Schupf1,3,Wayne Silverman4
1
New York State Institute for Basic Research in Developmental Disabilities,2The Taub Institute for
Research on Alzheimer’s Disease and the Aging Brain, Columbia University College of Physicians
and Surgeons,3Columbia University, G. H. Sergievski Center,4Kennedy Krieger Institute and Johns
Hopkins University School of Medicine
“Mild Cognitive Impairment” and Behavioral Concerns in Adults with Down Syndrome
Tiina K. Urv1,Sharon Krinsky-McHale2,Warren Zigman2,Wayne Silverman3
1
University of Massachusetts Medical School–Shriver Center,2New York State Institute for Basic
Research in Developmental Disabilities,3Kennedy Krieger Institute and Johns Hopkins University
School of Medicine
Are Cholinesterase Inhibitors Effective for Adults with Down Syndrome?
Wayne Silverman1,Warren Zigman2 ,Nicole Schupf2,3,Tiina Urv4,Robert Ryan2
1
Kennedy Krieger Institute and Johns Hopkins University School of Medicine,2New York State
Institute for Basic Research in Developmental Disabilities,3Columbia University, G. H. Sergievski
Center,4University of Massachusetts Medical School–Shriver Center
30
SYMPOSIUM 2
β42, Incident Dementia and Mortality in
Elevated Plasma Amyloid β-peptide Aβ
Down Syndrome
Nicole Schupf1, Bindu P. Patel2, Deborah Pang2, Warren B. Zigman3, Pankaj D. Mehta3,
Richard P. Mayeux2, Wayne Silverman4,
1
Columbia University Medical Center, New York, NY, USA; 2Columbia University Medical Center, New York,
USA; 3NYS Institute for Research on Developmental Disabilities, Staten Island, New York, 4Kennedy
Krieger Institute and Johns Hopkins School of Medicine, Baltimore, MD, USA.
Contact: Nicole Schupf, [email protected]
Background: Deposition of the amyloid beta peptide, β42, is thought to be an important initial step in the
pathogenesis of Alzheimer’s disease (AD). Individuals with Down syndrome (DS) have both increased
levels of β peptides and increased risk for Alzheimer’s disease.
Objectives: To examine the relation of plasma levels of β42 and β40 to risk of dementia in nondemented
adults with DS and to all-cause mortality.
Methods: Plasma β42 and β40 levels were measured at initial examination for 204 adults with DS.
Participants were assessed for cognitive and functional abilities, behavioral/ psychiatric conditions and
health status at 14-18 months over four cycles of data collection. Plasma levels of β42 and β40 levels
were measured using a combination of monoclonal antibody 6E10 (specific to an epitope present on 1-16
amino acid residues of β) and rabbit antisera R165 (vs. β42) and R162 (vs. β40) in a double antibody
sandwich ELISA.
Results: Among participants who were nondemented at baseline, those in the two highest tertiles of
plasma β42 levels were more than two times as likely to develop AD as those in the lowest tertile.
Compared with participants without AD, participants with prevalent AD had higher levels of plasma β42,
but did not differ in levels of plasma β40. Among all participants, those in the highest tertile of plasma β42
at baseline were two and one-half as likely to die over the study period as those in the lowest two tertiles.
Conclusions: Elevation in plasma β42 peptide level may serve as a biological marker sensitive to the
development and progression of AD and to risk of death. Our findings also support the hypothesis that
individual differences in β processing or deposition, distinct from more general overexpression of APP in
Down syndrome, may act as an initial step in the pathogenesis of AD.
Supported by New York State trough its Office of Mental Retardation and Developmental Disabilities, as
well as by Grants RO1 AG014763 (from the National Institute on Aging) and PO1 HD035897 & RO1
HD037425 (from the MR/DD Branch of the National Institute of Child Health and Human Development).
31
SYMPOSIUM 2
Apolipoprotein ε4 is Associated with Increased Mortality Risk in Nondemented
Adults with Down Syndrome
1
Warren B. Zigman1, Edmund C. Jenkins1, Benjamin Tycko2, Nicole Schupf1,3, Wayne Silverman4
New York State Institute for Basic Research in Developmental Disabilities, 1050 Forest Hill Road, Staten
Island, New York 10314. 2The Taub Institute for Research on Alzheimer’s Disease and the Aging Brain,
Columbia University College of Physicians and Surgeons, New York, NY. 3 Columbia University, G. H.
Sergievski Center, New York, NY. 4Kennedy Krieger Institute and Johns Hopkins University School of
Medicine, Baltimore, MD.
Contact e-mail: Warren Zigman, [email protected]
Down syndrome is characterized by increased mortality rates, both during early and later stages of life.
Causes of increased mortality early in life are primarily due to an increased incidence rate of congenital
heart disease, while causes of higher mortality rates later in life may be due to a number of factors,
including Alzheimer’s disease (AD) and a more general tendency toward premature aging. A welldocumented relationship has been noted between the occurrence of sporadic and/or familial late onset
Alzheimer’s disease and the gene for apolipoprotein E (APOE), encoded for by three alleles (APOE ε2, ε3,
and ε4) and located on chromosome 19. In the typically developing population and in adults with Down
syndrome, a relationship has been noted between the occurrence of late onset AD and APOE; the APOE
ε4 genotype has been associated with greater accumulation of beta-amyloid protein in the elderly with and
without Down syndrome and with a significantly increased age-specific risk for AD.
APOE genotype also has been related to survival in the general population, but its strong association with
AD makes interpretation of findings difficult. Previous studies of adults with Down syndrome have
consistently found that the presence of the APOE ε2 allele increases longevity and reduces the risk of
dementia, while the APOE ε4 allele increases risk for dementia. In contrast, reduced frequencies of the
APOE ε4 allele among elderly groups have been reported, suggesting that the APOE ε4 allele may be
associated with early mortality in this population. To disentangle effects of dementia from those of aging,
we compared mortality risk as a function of APOE genotype in 146 nondemented adults with Down
syndrome in a prospective study. We computed cumulative survival probability using Kaplan-Meier
survival analysis and Cox proportional hazards modeling to evaluate the association between APOE
genotype status and mortality.
Individuals with at least one APOE ε4 allele were approximately five times more likely to die within a 5- to
7-year study follow-up period than those without an APOE ε4 allele, adjusting for age at entry into the
study, sex, body mass index, level of mental retardation, and cholesterol level (Hazard Rate = 5.25, p =
.045, 95% CI: 1.04, 26.48). These results suggest that the APOE E ε4 allele has an independent and
strong relation to early mortality. Whether there are gene-environment or gene-gene interactions that are
responsible for these findings remain to be determined. Clearly, though, these results imply that APOE
plays an important and relatively direct role in determining longevity.
(Supported by New York State through its OMRDD, and by Grants P01 HD035897, R01 HD037425 and
R01 AG014673 from NIA and NICHD.)
32
SYMPOSIUM 2
“Mild Cognitive Impairment” and Behavioral Concerns in Adults with Down Syndrome
Tiina K. Urv1, Sharon Krinsky-McHale2, Warren Zigman2, & Wayne Silverman3
1
University of Massachusetts Medical School – Shriver Center, 200 Trapelo Road, Waltham, MA 02452.
2
New York State Institute for Basic Research in Developmental Disabilities. 3 Kennedy Krieger Institute and
Johns Hopkins University School of Medicine, Baltimore, MD.
Contact e-mail: Tiina Urv, [email protected]
Individuals with Down syndrome (DS) are at increased risk for developing Alzheimer’s disease, but there is
wide variation in age at onset of dementia. It is therefore imperative to identify those individuals who are at
greater and lesser risk for progressing to clinical dementia to enable individuals to receive potential
treatment and allow caregivers to develop long-term care plans.
In the general population, mild cognitive impairment (MCI) is characterized by cognitive decline of
insufficient severity for a diagnosis of dementia. It has been proposed to be a transitional state between
normal aging and early dementia, with individuals having MCI converting to clinical dementia at rates of
10% - 15% per year. The criteria for MCI in the general population include: memory complaints, objective
memory impairment, normal general cognitive functioning, and no significant declines in daily living skills.
While not part of the diagnostically defining features, MCI is associated with increased risk of neuropsychiatric symptoms.
Changes characteristic of MCI can be difficult to identify in any population having pre-existing cognitive
impairments, and differences between MCI and normal age-associated declines in cognition can be
extremely subtle. This study employed an operational definition of MCI appropriate for the population with
DS (MCI-DS) to examine behavior problems that become apparent to caregivers as declines in memory
begin but before frank dementia is present. We examined changes in the presentation of maladaptive
behaviors (using the Reiss Screen) in 72 adults with DS [14 males (Mean age = 51, SD = 4; Mean IQ =
39, SD = 7) and 58 females (Mean age = 51, SD = 6; Mean IQ = 38, SD = 7)]. These individuals were
divided into two groups. Adults with MCI-DS (n=27) experienced a decline of 15% or more on our Selective
Reminding Test of episodic memory but showed stability in adaptive behavior (declined less than 15%)
during a 14-18 month interval. A comparison group (n=45) displayed stability in both selective reminding
and adaptive behavior during a 14-18 month interval. [For inclusion, participants had to have a baseline
Selective Reminding score well above floor (> 20 on a scale from 0 to 48).]
Each of the 38 maladaptive behaviors in the Reiss Screen was categorized as “stable/getting better” or
“getting worse” over the 14-18 month interval, and groups were compared employing a series of 2 X 2 chisquare analyses. Participants in the “MCI-DS” group were significantly more likely to exhibit change in the
severity of tantrums (2.3% vs 29.6%), hostile behavior (4.5% vs 22.2%), and delusions (0 vs 7.4%), 4.99
d” x2 (1, N = 72) d” 11.31, .001 < ps <.05), suggesting that some specific concerns in maladaptive behavior
emerge in conjunction with relatively modest changes in cognitive functioning.
Additional chi-square analyses examined the subsequent dementia status within these Groups. A greater
proportion of individuals in the group with MCI-DS progressed to dementia (2.2% vs 20%), x2(1) = 8.5, p <
.01. A preliminary descriptive examination of the three significant behaviors distinguishing between the
Groups indicated that individuals displaying changes in hostile behavior and delusions were more likely to
develop dementia than were other people. This suggests that change in specific maladaptive behaviors
may be useful as “red flags,” alerting caregivers to the presence of possible declines in cognition that
might otherwise go unrecognized.
Supported by New York State through its OMRDD, and by Grants P01 HD035897 (Silverman), R01
HD037425 (Zigman) and R01 AG014673 (Schupf) from the NICHD (MR/DD Branch) and NIA.
33
SYMPOSIUM 2
Are Cholinesterase Inhibitors Effective for Adults with Down Syndrome?
1
Wayne Silverman1, Warren Zigman2, Nicole Schupf2,3, Tiina Urv4, and Robert Ryan2.
Kennedy Krieger Institute and Johns Hopkins University School of Medicine, 707 North Broadway, Suite
222s, Baltimore, MD 21205, USA. 2New York State Institute for Basic Research in Developmental
Disabilities, Staten Island, New York. 3Columbia University, G. H. Sergievski Center, New York, NY.
4
University of Massachusetts Medical School – Shriver Center, Waltham, MA.
Contact e-mail: Wayne Silverman, [email protected]
Alzheimer’s disease (AD) is the most prevalent cause of progressive dementia in old age, and risk is
dramatically increased among adults with Down syndrome (DS). No treatments targeting the underlying
pathology are currently available, but cholinesterase inhibitors are frequently prescribed to slow the rate of
declines in cognition and function. Studies of efficacy have produced mixed results in both the general
and DS populations, but there is general agreement that dramatic improvements in status are not to be
expected, and that tolerance to side effects has varied among individuals.
We have been conducting a large, prospective study of aging and dementia among adults with DS 45
years of age and older. Data were available on two separate occasions 14-18 months apart for 25 women
and 9 men taking Aricept, the most commonly prescribed cholinesterase inhibitor. These individuals, along
with 188 peers with DS not taking a cholinesterase inhibitor (141 women and 47 men), were divided into
four groups based upon dementia status: (a) No Dementia, (b) Questionable, meaning that some declines
have occurred but of insufficient severity to meet criteria for dementia, (c) Possible Dementia, and (d)
Definite Dementia.
Assessments of cognition included tests of episodic memory, verbal fluency, figure copying, block design,
and two separate tests of mental status [as described in Silverman, et al. (2004). AJMR, 109, 111-125].
Informant interviews included the AAMD Adaptive Behavior Scale (1974) and the Dementia Questionnaire
for Persons with Mental Retardation (DMR - Evenhuis, 1995). Informants were most likely aware of
medication status, but staff conducting assessments were not.
Results were analyzed employing Group (Aricept/Control) by Time by Dementia Status analyses of
variance. No 3-way interactions were significant, and Group by Time interactions failed to approach
signficance (p > .15) for most measures. Three Group by Time interactions were significant, however: (a)
total recall for our Selective Reminding test of episodic memory, F(1,130) = 7.3, p < .01, (b) the IBR
Mental Status Exam, F(1,137) = 16.4, p < .0001, and (c) the DS Mental Status Exam (Haxby, 1989),
F(1,147) = 4.3, p < .05. (Degrees of freedom varied because not all participants completed all tests.) All
three interactions were due to greater declines in the group taking Aricept. Taken together, results provide
fairly convincing evidence that this medication lacks efficacy, at least for adults with DS, even when its side
effects do not preclude use.
(Supported by N.Y.S. through its Office of Mental Retardation and Developmental Disabilities, and NIH
grants PO1 HD035897, RO1 HD037425, and R01 AG014673.)
34
Symposium 3
S Y M P O S I U M
3
Siblings of Individuals with Developmental Disabilities Across
the Life Span
Chair:
Ann Kaiser
Vanderbilt University
Discussant: Gael Orsmond
Boston University
35
SYMPOSIUM 3
Siblings of Individuals with Developmental Disabilities Across the Life Span
Chair:
Ann Kaiser, Vanderbilt University
Discussant: Gael Orsmond, Boston University
Adult Siblings of Persons with Disabilities: Findings from a National Survey
Richard C. Urbano & Robert M. Hodapp
1
Vanderbilt University
Sibling Relationships and Psychological Well-Being in Adults with and Without Siblings
with Intellectual Disabilities
Richard Hastings1, Mairead Doody1, Sarah O’Neill2 & Ian Grey2
1
University of Wales Bangor
2
Trinity College Dublin
Parent – Sibling Communication in Families of Children with Autism, Down Syndrome, and
Sickle Cell Disease
J. Carolyn Graff
University of Tennessee Health Science Center
36
SYMPOSIUM 3
Adult Siblings of Persons with Disabilities: Findings from a National Survey
Richard C. Urbano, & Robert M. Hodapp
Vanderbilt University
Although we know that most adult siblings are doing well and have reasonably good relationships with
their brother/sister with disabilities, studies generally involve the most involved siblings, and female as
opposed to male siblings. Little attention has generally been paid to etiology.
This study examines adult siblings and sibling relationships from a large-scale, national, web-based
survey. We were interested in: (1) female vs. male siblings, (2) close vs. less close siblings; and (3)
siblings of individuals with different types of disability.
Method: Adult siblings of 1,166 persons with disabilities participated in this study. Of these participants,
78.4% were female (910 F; 251 M) and 284 were siblings of persons with Down syndrome; 176 of persons
with autism; and 706 of persons with other forms of intellectual, physical, sensory, and emotional
disabilities (Other Disabilities group).
All participants answered an anonymous, web-based Adult Sibling Survey hosted by the Vanderbilt
Kennedy Center. The survey, which took about 25 minutes to complete, asked siblings about their and
their family’s demographics; their relationship and time spent with their brother/sister with disabilities;
health, depression, well-being, and rewards of being a sibling; and current and anticipated service needs.
Results: No gender differences emerged for the overall quality of the relationship between male and
female siblings, although two differences were noted. First, compared to U.S. Census norms, female (but
not male) siblings seemed to delay marriage and childbearing and were less likely to be divorced.
Compared to male siblings, females had more contact with their brother/sister with disabilities, t = 3.11, p
< .002, and reported that being a sibling to a brother/sister with disabilities has made them more
empathetic, understanding of differences, compassionate, and responsible (all p’s < .0001).
In families of 3 or more children, siblings who are closest, close but not closest, and not close with their
brother/sister with disabilities differed in the time spent in joint activities, F (2, 796) = 53.51, p < .0001, and
in a lessened sense of becoming more compassionate, empathetic, or responsible from having been a
sibling (p’s < .001).
Compared to siblings of persons with autism and with other disabilities, higher proportions of siblings of
persons with Down syndrome spent large amounts of time (5+ hrs./month)—and fewer spent little time (<2
hrs)—with their brother/sister with disabilities, ÷2 = 13.09, p < .011. Siblings of persons with Down
syndrome perceived themselves to have a closer, warmer relationship with their brother/sister, F (2, 1163)
= 18.18, p < .0001. Age-related changes differed by type of disability, with relationship quality decreasing
when the person with Down disabilities was aged 30+ and when the person with autism was 45+ (agegroup X disability, F (4, 1134) = 3.87, p < .004).
Conclusion: Although adult siblings of persons with disabilities remain an under-studied population, this
study expands our knowledge of their characteristics and relationships.
37
SYMPOSIUM 3
Sibling Relationships and Psychological Well-Being in Adults with and Without Siblings
with Intellectual Disabilities
Richard Hastings1, Mairead Doody1, Sarah O’Neill2 & Ian Grey2
1
University of Wales Bangor
2
Trinity College Dublin
Correspondence: Richard Hastings PhD, School of Psychology, University of Wales Bangor, Bangor,
Gwynedd, LL57 2AS, Wales, UK.
E-mail: [email protected]
Although there has been considerable research interest in younger siblings of children with developmental
disabilities, few researchers have studied the experiences of adult siblings of people with intellectual
disabilities. The main aims of the present study were: 1. To compare sibling psychological adjustment and
dimensions of the sibling relationship between adult siblings who either do or do not have a brother or
sister with intellectual disabilities, and 2. To explore correlates of sibling adjustment and relationships for
adult siblings of individuals with intellectual disabilities. An exploratory aim was to adapt a measure of
parental positive perceptions for siblings to establish its utility for future research. Participants were 123
adults who did not have a sibling with disability and 63 adults who had a sibling with intellectual disabilities.
Using an Internet survey in the UK and Ireland, siblings were asked to report on dimensions of their sibling
relationships as adults (contact with the sibling, warmth, conflict, and rivalry), and their own psychological
well-being. For a sub-sample, the emotional dimension of the sibling relationship was assessed using the
Five Minute Speech Sample measure of Expressed Emotion (specifically, Criticism). Participants with a
sibling with intellectual disability also completed an adapted version of the Positive Contributions Scale of
the Kansas Inventory of Parental Perceptions. Group comparisons showed no differences in rivalry,
conflict, criticism, or psychological well-being. Control participants were warmer, lived further away, and
saw their siblings less frequently than siblings of people with intellectual disabilities. Siblings of people with
intellectual disability were telephoned less frequently by their brother or sister. Using correlation and
regression analyses, a number of predictors of sibling relationship dimensions were identified for the
siblings of people with intellectual disabilities. Warmth was higher when the brother or sister had mild/
moderate as opposed to severe/profound intellectual disability, when the sibling also expressed more
positive perceptions, and when telephone contact was more frequent. Conflict was higher when the
siblings had poorer psychological well-being, were living alone, and were younger than their brother or
sister with intellectual disability. Finally, criticism and positive perceptions were negatively associated.
Although these data are consistent with much sibling research suggesting little evidence of negative
effects of people with intellectual disabilities on their siblings, there may be some subtle differences that
are worthy of exploration in future research. Some of these differences may be important to understand
perhaps in later life when siblings may become more involved as carers. Siblings also report considerable
positive perceptions of their brother or sister with intellectual disability, and these experiences need to be
more fully researched.
38
SYMPOSIUM 3
Parent – Sibling Communication in Families of Children with Autism, Down syndrome,
and Sickle Cell Disease
J. Carolyn Graff
University of Tennessee Health Science Center;
711 Jefferson Avenue, Memphis, TN 38105
Contact: [email protected]
Introduction: Integrating genomics, the translation of knowledge of the human genome into benefits for
children with genetic disorders and their family members, is critical to improving the health and well being
of these individuals. Researchers and professionals have focused on children with genetic disorders and
their parents and turn their attention to siblings when they are suspected of being at risk for or having a
genetic disorder or other problems. Although a non-categorical approach has been used to consider the
impact of a disorder on children and their families (Stein & Jessop, 1989), differences can be expected to
exist across genetic disorders. The purpose of this study is to describe the structure, content, and function
of parent-sibling communication about autism, Down syndrome (DS), and sickle cell disease (SCD);
describe factors within and outside the parent-sibling dyad that influence their communication; compare
and contrast parent responses from these groups; and consider the relevance of the non-categorical
approach for parent and sibling experiences.
Methods: Separate audio taped focus group interviews were held with parents of children with autism (n =
12) and DS (n = 10). Parents were recruited from local support groups and using snowball or network
sampling. Recruitment of parents of children with SCD is underway through the St. Jude Children’s
Research Hospital. SCD focus group interviews will be held next month. Transcribed data have been
entered into NVivo7 and coded using interpretational analysis (Tesch, 1990). Categories emerging from
coded data were analyzed further to identify themes. Themes and categories were examined in the
context of select factors from Fitzpatrick and Caughlin’s (2002) theoretical approaches to family
relationships.
Results: Parents of children with autism initiated communication with siblings to explain the child’s
behaviors, therapy, and need for siblings to help with therapy or chores. Siblings were frustrated with their
brother or sister’s behaviors and wanted more attention and to find something to help their brother or
sister. Unpredictable behaviors of children with autism influenced parent-sibling communication. In the DS
group, siblings initiated discussions with their parents to clarify the present health and educational status
of children with DS, learn more about DS, and discuss their own future and the future of children with DS.
Health problems of children with DS and the unknown future influenced parent-sibling communication.
Siblings in both groups were advocates and protectors of their brother or sister. Analysis of data from the
SCD focus group interviews will be included in the final presentation.
Discussion: Although parents and siblings in the autism and DS groups have similar experiences and use
similar strategies to understand and adjust to changes within their family, they experience different
challenges based on the behaviors, health, and functioning ability of the child with a genetic disorder.
Further exploration of these similarities and differences with siblings should be conducted to tailor sibling
and parent supports that build on existing individual and family strengths and resources.
Fitzpatrick, M.A., Caughlin, J.P. (2002). Interpersonal communication in family relationships: In
M.L. Knapp & J.A. Daly (Eds.), Handbook of interpersonal communication (3rd ed.) (pp. 726-777).
Thousand Oaks, CA: Sage.
Stein, R.E., & Jessop, D.L. (1989). What diagnosis does not tell: The case for a noncategorical
approach to chronic illness in childhood. Social Science and Medicine, 29(6), 769-778.
Tesch, R. (1990). Qualitative research: Analysis types and software tools. New York, NY:
Falmer Press.
39
Symposium 4
S Y M P O S I U M
4
Asian Families of Children with Developmental Disabilities
Chair:
Jin Y. Shin
Department of Psychology, Hofstra University
Discussant: Laraine M. Glidden
Center on Health and Education, Georgetown
University
40
SYMPOSIUM 4
Asian Families of Children with Developmental Disabilities
Chair:
Jin Y. Shin, Department of Psychology, Hofstra University
Discussant: Laraine M. Glidden, Center on Health and Education, Georgetown University
Agreement on Childhood Disability between Parents and Teachers in Vietnam
Jin Y. Shin,12 Nguyen Viet Nhan, 3 Stavros S. Valenti,1 Kathleen Crittenden,4 Michel Flory 2
1
Department of Psychology, Hofstra University
2
Infant Development, New York State Institute for Basic Research in Developmental Disabilities
3
Hue Medical College, Vietnam
4
University of Illinois at Chicago
A Comparison of Perspectives on Sibling Relationships Involving an Individual with Special Needs
Sun Young Ryu
Department of Health and Behavioral Studies, Teachers College, Columbia University
Parent Language Input and Its Impact on the Engagement of Children with Autism: The Chinese
Perspective
Peishi Wang
Graduate Program in Special Education, Department of Educational and Community Programs, Queens
College, City University of New York
41
SYMPOSIUM 4
Agreement on Childhood Disability between Parents and Teachers in Vietnam
Jin Y. Shin,1,2 Nguyen Viet Nhan, 3 Stavros S. Valenti,1 Kathleen Crittenden,4 Michel Flory 2
1
Department of Psychology, Hofstra University,
2
Infant Development, New York State Institute for Basic Research in Developmental Disabilities
3
Hue Medical College, Vietnam
4
University of Illinois at Chicago
Contact: [email protected]
135 Hofstra University, Hempstead, NY 11549
Introduction: This research examined the agreement on disability among the teachers and parents of
children with cognitive delays in Vietnam. While the diagnosis and classification of children with
disabilities are persistent concerns in all clinical and educational services, these concerns are magnified in
developing countries. Standardized diagnostic and assessment tools and systems either do not exist or
are not readily available in many developing countries. While we attempted to identify children with
cognitive delays with simple strategies that include screening procedures, we also documented the
discrepancies among parents and teachers in perceiving the level of disability in their children.
Methods: Teachers (n = 57) in kindergarten programs (for children aged 2 to 6) who identified children as
having cognitive delays, and the mothers (n = 106) and fathers (n = 93) of these children rated the child’s
level of disability based on the ABILITIES Index (Simeonsson & Bailey, 1988).
Results: The teachers rated the level of child’s functioning more severely, especially in the areas of
intellectual disabilities and behavior problems, than did mothers and fathers. There was more agreement
between mothers and fathers than between parents and teachers. The teachers and fathers agreed the
least in most areas of child’s level of functioning. Logistic regressions examining the factors that predicted
agreement or disagreement among parents and teachers revealed that when the child’s disability was
genetically related (e.g., Downs Syndrome) or physical (e.g., cerebral palsy) it was more likely that
teachers and parents agreed. Mothers and fathers were more likely to agree on the level of child’s
intellectual disability when their children were older.
Discussion: Agreement and disagreement on children’s intellectual and other disabilities is discussed in
the context of Vietnam. The lower agreement between teachers and parents could be due to lack of
information among the parents regarding intellectual and developmental disabilities and different beliefs
and stigma about having a child with disability.
42
SYMPOSIUM 4
A Comparison of Perspectives on Sibling Relationships
Involving an Individual with Special Needs
Sun Young Ryu
Department of Health and Behavior Studies
Teachers College, Columbia University
Contact: [email protected]
Introduction: Sibling relationships are one of the most long-lasting and enduring human relationships.
Sibling relationships involving an individual with special needs are critical for the well-being of siblings with
special needs because typically developing siblings are often named by the parent to be potential caregivers and sources of support for their siblings with special needs after parents’ death. However,
perspective differences associated with family roles and different cultures have not been documented in
terms of sibling relationships, family relationships, sibling self-concept, and sibling commitment.
Methods: The current study investigated perspectives on family relationships, sibling relationships, sibling
self-concept, and sibling commitment between typically developing siblings and their parents from
American and Korean cultural backgrounds. 50 American and 66 Korean young adults whose ages were
between 18 and 30 and who had at least one sibling with special needs, and one of their parents
participated in the study. Participants responded to self-administered questionnaires that measured the
four dependent variables.
Results: Sibling groups and parent groups reported significantly different perspectives for overall family
relationship and sibling commitment. Parents were more positive than their typically developing children
for family relationships, and typically developing siblings were more positive than their parents for sibling
commitment. American groups and Korean groups reported significantly different perspectives for overall
family relationship and sibling self-concept. The American groups were more positive than the Korean
groups for both family relationship and sibling self-concept. Especially on sibling commitment, American
siblings demonstrated present-focused and affective-oriented sibling commitment while Korean siblings
showed future-focused and instrumental-oriented sibling commitment. For example, about 40% of the
typically developing American siblings expected their siblings with special needs to live with them whereas
about 80% of typically developing Korean siblings expected their siblings with special needs to live with
them after their parents’ death. Some of the demographic variables were significantly related to the four
measures.
Discussion: Multi-role and multicultural perspectives on those measures can be useful in planning and
coordinating services such as guardianship, living arrangement, and long-term financial support for
individuals with special needs. Future research should examine in particular the role of age and gender in
sibling perspectives.
43
SYMPOSIUM 4
Parent Language Input and Its Impact on the Engagement of Children with Autism:
The Chinese Perspective
Peishi Wang
Graduate Program in Special Education,
Dept of Educational and Community Programs,
Queens College, City University of New York
Contact: [email protected]
Introduction: Research on parent-child interaction in the U.S. reported that children with autism tend to
demonstrate less compliance and more avoidance than children in the nonautistic group. Several studies
revealed that parents of children with autism used more attention getting behaviors, increased their
physical proximity, and used more nonverbal prompts than parents in the nonautistic group. Some
speculated that mothers of children with autism primarily used control behaviors to support and encourage
their child to participate in the interaction.
There is very little research done in the area of interactions between parents and children with autism in
the People’s Republic of China. The current study aims to explore the pattern of Chinese parents
language input when interacting with their children with autism and the impact it has on the engagement of
their children.
Methods: A pretest-posttest group design was implemented for this study. Twenty-seven parents of
children with autism in the Northeastern region of China were randomly assigned to either the training or
the control group. Parents received a total of twenty hours of training in a format that included both group
and individual sessions. The parent training program was based on principles and tactics derived from
applied behavior analysis, naturalistic teaching and the TEACCH philosophy. Parent-child interactions
were examined using videotaped observations at their homes and analyzed using the Kid Talk Code
(Kaiser et al., 2004).
Results: Analysis of this data set is ongoing. Information on the types of language input (such as
modeling, expansions, instructions, praise, etc) that parents provided and its impact on the child’s
engagement during a free play interaction will be presented. Group comparisons will be made between pre
and post tests on the types of language input and children’s engagement behavior.
Discussion: Results from this study will increase our understanding of the patterns of Chinese parentchild interactions. Implications for future parent training programs will be highlighted.
44
Symposium 5
S Y M P O S I U M
5
Children with Down Syndrome: Parent and Child Supports
Chair:
Mary Ann Romski
Georgia State University
Discussant: Nancy Brady
University of Kansas, Lawrence,Kansas
45
SYMPOSIUM 5
Children with Down Syndrome: Parent and Child Supports
Chair:
Mary Ann Romski, Georgia State University
Discussant: Nancy Brady, University of Kansas, Lawrence, Kansas
Parent-Child Communication at Home in Preschool Children with Down Syndrome and
Autism Spectrum Disorders
Ann P. Kaiser, Terry B. Hancock and Prathibha Vijay
Vanderbilt University, Nashville, TN
Coping with Frustration in Children with Down Syndrome
Laudan B. Jahromia, Amanda Gulsrudb, and Connie Kasarib
a
Arizona State University & bUniversity of California, Los Angeles
Examining Parental Stress and Parental Perceptions of Toddler’s Communication
Development Following Language Intervention: A Comparison between Children
with and without Down Syndrome
Ashlyn L. Smith and Mary Ann Romski
Georgia State University
46
SYMPOSIUM 5
Parent-Child Communication at Home in Preschool Children with Down Syndrome
and Autism Spectrum Disorders
Ann P. Kaiser, Terry B. Hancock and Prathibha Vijay
Vanderbilt University
Correspondence: Ann P. Kaiser, PhD, Vanderbilt Kennedy Center,
Vanderbilt University, Nashville, TN 37203 Email: [email protected]
Both young children with Down syndrome (DS) and young children with autism spectrum disorders (ASD)
are delayed in their development of spoken language, however, they differ in their social communication
skills. Children with DS are typically socially responsive in early communicative interaction, but show
increasing delays in productive language skill relative to age over the preschool years. In contrast, children
with ASD have difficulty with joint attention, and are less responsive in early communicative interactions.
While tested differences in children’s language are informative, observing children’s communication and
the effects their communication has on their parents can provide additional information about the impact of
social communicative abilities associated with specific disabilities on children’s learning environments.
The current study examined: a) differences in children’s communication and behavior in DS and ASD
children; b) differences in the responsiveness of their parents to children’s communication attempts; and c)
differences in parental language input to children in the two groups during everyday communication
interaction. .
Method. Children with DS (N=15) and ASD (N=15) and their parents were observed during 30 minute toyplay based interactions at home. The two groups were matched for gender (4 females; 11 males/group),
age (Mn =42 mo) and MLU (Mn = 1.50). The majority of parents in both groups were Euro-American
mothers with at least a high school education. Data were transcribed from video tapes and coded using a
Procoder based protocol. Inter-observer agreement on parent and child behaviors was calculated for 50%
of the coded observations.
Results. As expected due to matching, children in the two groups did not differ significantly in MLU or
tested language, however, they differed significantly on most measures of productive language use.
Children with DS had nearly twice as many spontaneous utterances; used more diverse words, more
diverse utterances, and took more verbal turns than children with ASD. The two groups of children did not
differ in terms of their observed negative behavior. Parents in both groups were highly responsive and
positive, responding to more than 60% of all child utterances, and providing about l positive statement per
minute. Because children with DS talked more, their parents provided significantly more responsive
language models. Parent of children with DS had significantly higher MLU’s than parents of ASD children,
even though child MLU was similar in the two groups. These findings suggest that the social
communicative aspects of children’s language use affect both the quantity and the quality of parent input.
Follow up qualitative analyses focus on the pragmatic functions of parent and child communicative
utterances.
47
SYMPOSIUM 5
Coping with Frustration in Children with Down Syndrome
a
Laudan B. Jahromia, Amanda Gulsrudb, & Connie Kasarib
Arizona State University & bUniversity of California, Los Angeles
Box 951521, 3132B MH, Los Angeles, CA 90095-1521
Contact: [email protected]
Previous research has highlighted the salience of looking behavior and eye-contact in the behavioral
repertoire of children with Down syndrome (e.g., Kasari, Freeman, Mundy, & Sigman, 1995). This study
explored the co-occurrence of these looking behaviors and assistance seeking during a task designed to
elicit frustration in children with Down syndrome, nonspecific mental retardation and typical development.
Participants/Procedures: Participants included 63 children (19 children with Down syndrome, 22 children
with nonspecific mental retardation, and 22 children with typical development). Children were matched for
mental age (average of 52.5 months) and both groups of children with mental retardation were matched
for chronological age (average of 82 months). Unsolvable puzzles were administered to the children (e.g.,
Smiley & Dweck, 1994). Negativity/frustration and the coping strategies of assistance seeking and
experimenter orientation were coded during the puzzle task using the Laboratory Temperament
Assessment Battery (Goldsmith et al., 1999).
Results: Group effects were found for negativity, assistance seeking with and without experimenter
orientation, and experimenter orientation without assistance seeking. Children with Down syndrome
displayed negativity for a greater proportion of time (M = .22) than did children with mental retardation (M
= .11), d = 4.38 and marginally more than those with typical development (M = .15; p =.06), d = 2.79.
There was no significant difference in overall negativity in the latter two groups. For experimenter
orientation with and without assistance seeking, typical children engaged in these behavioral combinations
for a significantly greater proportion of time (M = .16 and M=.21) than did children with mental retardation
(M = .04, M=06). There was a marginal group difference in experimenter orientation with no assistance
seeking, F (2, 62) = 2.82, p < .07. Children with Down syndrome used this behavioral combination for a
greater proportion of time (M = .22) than typical children (M = .10), d = .76.
A sequential methods approach was used to explore the associations between negativity and coping
strategy, specifically to understand the relation between specific regulation strategies and decreases in
children’s negativity. Interestingly, the results of these analyses for children with Down syndrome and
mental retardation revealed that no behavior or behavioral combination showed a significant cooccurrence with decreasing negativity. For typical children, a significant sequential contingency was found
for experimenter orientation with assistance seeking. This behavioral combination co-occurred with
decreasing negativity more often than chance for a greater number of typical children.
Conclusion: These results suggest that although children with Down syndrome and mental retardation
are displaying increased negativity during a frustrating task they are not systematically using an effective
coping strategy (assistance seeking) to decrease the negativity such as children with typical development.
Implications for treatment will be discussed.
Goldsmith, H. H., Reilly, J., Lemery, K. S., Longley, S., & Prescott, A. (1999). The Laboratory
Temperament Assessment Battery: Preschool Version. University of Wisconsin.
Kasari, C., Freeman, S., Mundy, P., & Sigman, M. D. (1995). Attention regulation by children with Down
syndrome: Coordinated joint attention and social referencing looks. American Journal on Mental
Retardation, 100, 128-136.
Smiley, P. A., & Dweck, C. S. (1994). Individual differences in achievement goals among young children.
Child Development, 65, 1723-1743.
48
SYMPOSIUM 5
Examining Parental Stress and Parental Perceptions of Toddler’s Communication
Development Following Language Intervention: A Comparison between
Children with and without Down Syndrome
Ashlyn L. Smith and Mary Ann Romski
Georgia State University, Atlanta, Georgia
Contact: [email protected]
Children with developmental disabilities encompass a wide variety of diagnoses including but not limited to
autism, Down syndrome, and Fragile X syndrome. Research has shown that the child’s diagnosis can
have differing impacts on parent stress. Specifically, parents of children with Down syndrome report lower
levels of child-related stress (Kasari & Sigman, 1997; Hodapp, Ricci, Ly, and Fidler, 2003). The greater
sociability and adaptive functioning of these children appears to lead to lower levels of perceived stress by
the parent. There have been few studies however, that investigate the role that a child’s communication
ability has on parental stress. The communication abilities of children with disabilities are often delayed,
and in some children are significantly impaired. Interventions focused on improving the communication
abilities of these children are often implemented without parent involvement even though the transactional
nature of communication suggests that parent involvement will result in more positive child outcomes.
Involving parents in language interventions is critical in providing not only positive outcomes for children
but for the psychological well-being of parents as well. Research in the area of parent-implemented
interventions has shown that teaching parents new skills to support their child’s language development
can in fact improve developmental outcomes and reduce problem behavior. This paper examines the
differential effects of a parent-implemented language intervention on parent stress and parent perceptions
of their child’s language development. It compares a sample of parents of children with and without Down
syndrome and is part of a larger study comparing the effects of three different parent implemented
language interventions on the communication development of toddlers who encounter significant difficulty
acquiring speech.
60 toddlers and their parents were recruited for this study. 18 toddlers were diagnosed with Down
syndrome with a mean age of 28.61 months (SD = 3.13). 42 were diagnosed with other etiologies, such
as, pervasive developmental disorder (PDD), cerebral palsy, or other genetic disorders, with a mean age
of 29.90 (SD = 5.04). Participants were randomly assigned to one of three parent-implemented language
interventions: Spoken Communication Intervention (SCI), which focused on spoken interaction, or one of
2 augmented language interventions, Augmented Communication Input Intervention (ACI), and
Augmented Communication Output Intervention (ACO). Language skills, measures of parental stress and
parent perceptions of language development, were all measured at pre and post intervention.
Results indicate that parents of children with Down syndrome showed significantly lower levels of total
parent stress at baseline as compared to parents of children with other etiologies. Following language
intervention, parents of children with Down syndrome also decreased significantly on the total stress scale
and the parent distress subscale of the PSI, while parents of children with other etiologies did not. Results
also show that parent’s perceptions about their success in impacting their child’s communication
development were not significantly different at baseline. However, parent’s feelings of success increased
significantly for parents of children with other etiologies, but not for parents of children with Down
syndrome.
This study supports previous research that parents of children with Down syndrome exhibit a lower stress
profile than parents of children with other etiologies. The findings also suggest that participating in a
parent implemented language intervention serves to further reduce parent stress in parents of children
with Down syndrome.
49
Symposium 6
S Y M P O S I U M
6
Down Syndrome: Gene Expression and Beyond
Chair:
Benjamin Tycko
Institute for Cancer Genetics,
Herbert Irving Cancer Research Center
Discussant: Nicole Schupf
Taub Institute for Research on Alzheimer’s Disease
and the Aging Brain, Columbia University Medical
Center, and NYS Institute for Basic Research in
Developmental Disabilities
50
SYMPOSIUM 6
Down Syndrome: Gene Expression and Beyond
Chair:
Benjamin Tycko, Institute for Cancer Genetics, Herbert Irving Cancer Research
Center
Discussant: Nicole Schupf, Taub Institute for Research on Alzheimer’s Disease and the Aging
Brain, Columbia University Medical Center, and NYS Institute for Basic Research in
Developmental Disabilities
Function of Chromosome 21 Gene Products Investigated in BAC-Transgenic Mice
Luzhou Xing, Martha Salas, Chyuan-Sheng Lin, Ning Kon, Muyang Li, Wei
Gu, Warren Zigman, Wayne Silverman and Benjamin Tycko
Department of Pathology, Taub Institute and Institute for Cancer Genetics, Columbia University
Medical Center, New York NY, Department of Psychology, New York State Institute for Basic
Research, New York, NY, and Department of Behavioral Psychology, Kennedy Krieger Institute,
Baltimore, MD
Role of PIP2 Phosphatase Synaptojanin 1 in Down Syndrome-Linked Brain Dysfunction
Sergey V. Voronov, Samuel G. Frere, Silvia Giovedi, Elizabeth A. Pollina, Cecilia Schmidt, Laurent
Cimasoni, Christelle Borel, Markus R. Wenk, Stylianos E. Antonarakis, Katheleen Gardiner, Muriel
T. Davisson, Ottavio Arancio, Pietro De Camilli, Gilbert Di Paolo
Taub Institute for Research on Alzheimer’s Disease and the Aging Brain,
Columbia University Medical Center
Consequences of Over-Expression of Mnb/Dyrk1A in Down Syndrome
Jerzy Wegiel*, Karol W. Dowjat, Wojciech Kaczmarski, Yu-Wen Hwang, Cheng-Xin Gong, Tatyana
Adayev, Madhabi Barua, Izabela Kuchna, Krzysztof Nowicki, Jarek Wegiel, Humi Imaki, Pankaj
Mehta, Arthur Dalton, Georgio Albertini, Ausma Rabe, Zhihou Liang, Fei Liu, Khalid Iqbal, Inge
Grundke-Iqbal, Noriko Murakami, Naryan Ramakrishna, Wayne P. Silverman
New York State Institute for Basic Research in Developmental Disabilities, and Kennedy Krieger
Institute, Johns Hopkins University School of Medicine
51
SYMPOSIUM 6
Function of Chromosome 21 Gene Products Investigated in BAC-Transgenic Mice
Luzhou Xing, Martha Salas, Chyuan-Sheng Lin, Ning Kon, Muyang Li, Wei
Gu, Warren Zigman, Wayne Silverman and Benjamin Tycko*
Department of Pathology, Taub Institute and Institute for Cancer Genetics, Columbia University Medical
Center, New York NY, Department of Psychology, New York State Institute for Basic Research, New York,
NY, and Department of Behavioral Psychology, Kennedy Krieger Institute, Baltimore, MD.
Contact: Benjamin Tycko, M.D., Ph.D., Herbert Irving Cancer Research Center
1130 St. Nicholas Avenue, New York, NY 10032;[email protected]
Introduction: The COL6A1 gene, in chromosome band 21q22.3, encodes a subunit of Type VI collagen
and has been a candidate for contributing to cardiac and skin abnormalities in Down syndrome. The
DSCR1 gene, in chromosome band 21q22.12, encodes an inhibitor of calcineurin and has been a
candidate for contributing to several phenotypes in Down syndrome, including cardiac and cognitive
defects, as well as resistance against solid tumors. We have started to study these 2 genes by creating
bacterial artificial chromosome (BAC)-transgenic mice over-expressing each of them. As an experimental
approach, BAC transgenic mice have potential advantages including appropriate tissue-specific
expression and copy number-dependent expression of the transgenes.
Material and methods: First, to investigate the effects of COL6A1 over-expression on cardiac and skin
morphology, we created transgenic mice with genomic integrations of a BAC containing the human
COL6A1 gene with 82 kb of upstream and 62 kb of downstream DNA. We characterized 2 transgenic
lines, one low-copy and the other high-copy for the COL6A1 transgene. Second, to investigate the
biochemistry and function of the DSCR1 protein in vivo we created BAC-transgenic mice as an allelic
series with progressively increasing dosage of this gene. The DSCR1 BAC was engineered with a tandem
tag to facilitate purification and characterization of high molecular weight DSCR1 protein complexes from
native tissues of these mice.
Results:
1. COL6A1: In high-copy and low-copy transgenic lines, we found correct temporal and spatial expression
of COL6A1 mRNA, paralleling the expression of the endogenous murine Col6a1 gene in a panel of 9 adult
and 4 fetal organs. In this panel the only exception to faithful expression was the fetal lung, in which the
transgene was expressed at disproportionately low levels compared to the endogenous gene. Expression
of COL6A1 mRNA from the BAC transgene was copy-number-dependent, and the increased gene dosage
correlated with increased production of alpha (VI) collagen in skin and heart, as indicated by western
blotting and immunohistochemistry. The low-copy and high-copy COL6A1 BAC-transgenic mice were born
and survived in normal Mendelian proportions, and we did not observe cardiac malformations or altered
skin histology in these animals. These data indicate that the major promoter and enhancer sequences
regulating COL6A1 expression are present in this 167 kb BAC clone. The lack of an obvious cardiac or
skin phenotype in the COL6A1 BAC-transgenic mice suggests that the increased copy number of this
gene does not, by itself, account for these phenotypes in DS.
2. DSCR1: By western blotting we verified copy-number-dependent over-expression of DSCR1 protein
dependent on transgene dosage in our allelic series of DSCR1-BAC transgenic mice. Initial phenotypic
and biochemical findings in these mice will be discussed, with the objective of better understanding the
cognitive and cardiac pathophysiology of Down syndrome.
52
SYMPOSIUM 6
Role of PIP2 Phosphatase Synaptojanin 1 in Down Syndrome-Linked Brain Dysfunction
Sergey V. Voronov, Samuel G. Frere, Silvia Giovedi, Elizabeth A. Pollina, Cecilia Schmidt, Laurent
Cimasoni, Christelle Borel, Markus R. Wenk, Stylianos E. Antonarakis, Katheleen Gardiner, Muriel T.
Davisson, Ottavio Arancio, Pietro De Camilli, Gilbert Di Paolo*
Contact: Gilbert Di Paolo, Department of Pathology and Cell Biology, Taub Institute for Research on
Alzheimer’s Disease and the Aging Brain, Columbia University, New York, NY 10032;
[email protected]
Introduction: In DS, integrated gene expression is altered due to the presence of a third copy of human
chromosome 21 (HC21). Although mental retardation has been linked to non-overlapping regions of HC21,
indicating the multigenicity of its etiology, the relative contribution of single genes to this phenotype is unknown.
Here we propose that Synj1 is a strong candidate for contributions to mental retardation in DS. Synj1 encodes
synaptojanin 1, a brain- enriched lipid phosphatase that dephosphorylates phosphatidylinositol-4,5bisphosphate (PIP2). This lipid regulates a myriad of biological functions, including signal transduction,
membrane trafficking and ion channels. Our previous work has provided strong evidence that PIP2 regulates
synaptic vesicle trafficking and thereby, neurotransmission. This property is largely due to the ability of this lipid
to recruit to the plasma membrane key cytosolic components of the endocytic machinery, which mediates the
recycling of synaptic vesicles. Our hypothesis is that Synj1-linked PIP2 metabolism imbalance in DS may
interfere with normal synaptic physiology and contribute to behavioral deficits in DS.
Material and Methods: To address the role of Synj1 in DS, we have used the following genetic tools: (i)
segmentally trisomic mice Ts65Dn; (ii) BAC transgenic mice containing one extra copy of Synj1
[Tg(Synj1)]; and (iii) Synj1+/- mice. Our lipid biochemistry experiments have involved PIP2 phosphatase
assays from brain extracts and mass measurements. For behavioral analyses, we have used the Morris
water maze paradigm and variants thereof.
Results:
1. Evidence for gene dosage imbalance for Synj in DS mouse models. We have performed a Western
Blot analysis using brain homogenates from wild-type, Ts65Dn mice and two lines of transgenic mice
overexpressing Synj1. We found a comparable increase (40-60%) in the levels of synaptojanin 1 in
Ts65Dn and Tg(Synj1) mice relative to controls. This increase reflects the presence of three copies of the
gene encoding synaptojanin 1, Synj1. Importantly, Synj1 overexpression was fully rescued by genetically
restoring a normal Synj1 copy number in Ts65Dn mice (Ts65Dn/Synj1+/+/-), which was achieved by
breeding Ts65Dn mice with Synj1+/- (targeted mutation) animals.
2. Evidence for PIP2 metabolism defects in DS mouse models. Using a PIP2 phosphatase assay, we
found a 33%±10 and a 54%±19 increase in the production of PIP with Ts65Dn and Tg(Synj1) cytosol,
respectively, relative to controls, indicating that the overall PIP2 phosphatase activity of mutant is
significantly enhanced as a result of Synj1 triplication. Importantly, the PIP2 phosphatase activity of
Ts65Dn/Synj1+/+/- cytosol was comparable to that of WT brains. We also found that a PIP2 deficiency is
present in the brain of Ts65Dn mice and that the underlying cause is trisomy of the Synj1 gene. Although
this deficiency may appear to be subtle, it is likely to be meaningful from a neurophysiological point of
view, as PIP2 metabolism, like that of cholesterol, is subject to a tight homeostatic regulation.
3. Evidence for learning deficits in transgenic mice overexpressing Synj1. We next tested whether Synj1
overexpression affects the learning ability of Tg(Synj1) mice. We have begun our studies using the Morris
water maze paradigm. In our first two independent sets of experiments, we have analyzed a total of 29
animals [16 control mice (5 males and 11 females) and 13 transgenic mice (4 males and 9 females), age:
3-4 months]. We found that animals from both genotypes performed very well in the visible platform test,
where they significantly improved their performance over the course of four sessions (one session/day).
Similarly, there was no obvious difference in the escape latency between control and Tg(Synj1) animals in
the hidden platform test. However, control animals showed clear signs that they had (at least partially)
learned the task, since they spent significantly more time in the TQ quadrant compared to the other three
quadrants (p < 0.001 for opposite quadrant and p < 0.05 for the adjacent quadrants according to ANOVA’s
Bonferroni multiple test comparison). On the contrary, Tg(Synj1) animals did not show any signs of
learning, as they spent a comparable fraction of their swimming time in all four quadrants. In conclusion,
our experiments indicate that Tg(Synj1) mice exhibit learning deficits.
53
SYMPOSIUM 6
Consequences of Over-Expression of Mnb/Dyrk1A in Down Syndrome
Jerzy Wegiel, Karol W. Dowjat, Wojciech Kaczmarski, Yu-Wen Hwang, Cheng-Xin Gong, Tatyana Adayev,
Madhabi Barua, Izabela Kuchna, Krzysztof Nowicki, Jarek Wegiel, Humi Imaki, Pankaj Mehta, Arthur
Dalton, Georgio Albertini, Ausma Rabe, Zhihou Liang, Fei Liu, Khalid Iqbal, Inge Grundke-Iqbal, Noriko
Murakami, Naryan Ramakrishna, Wayne P. Silverman
New York State Institute for Basic Research in Developmental Disabilities, Staten Island NY and Kennedy
Krieger Institute, Johns Hopkins University School of Medicine, Baltimore, MD
Contact: Jerzy Wegiel, New York State Institute for Basic Research in Developmental Disabilities,
1050 Forest Hill Road, Staten Island NY 10314; [email protected]
Introduction: The gene coding the minibrain kinase/dual-specificity tyrosine phosphorylated and
regulated kinase 1A (Mnb/Dyrk1A) is located in the Down syndrome (DS) critical region of chromosome
21. The third copy of Mnb/Dyrk1A is believed to contribute to DS phenotype, although the mechanisms
and exact role of this kinase in developmental and age associated changes in DS are not defined. Current
study characterizes (1) level of over-expression of DYRK1A in DS brain, (2) different pattern of ageassociated decrease of levels of DYRK1A isoform 1 and 2, and (3) interaction with tau protein resulting in
modification of tau-hyperphosphorylation and contribution to neurofibrillary degeneration.
Material and Methods: Morphological and biochemical study of brains and other organs of DS, AD and
control subjects, Ts65Dn and control mice was focused on distribution and overexpression of DYRK1A
protein and its isoforms, and pathological consequences of phosphorylation of DYRK1A substrates.
Results:
1. Trisomy-driven overexpression of DYRK1A kinase in the brain of subjects with Down syndrome. In DS
infants the ratio of DYRK1A versus control subjects varied in range from 1.1 in the white matter, to 2.1 in
occipital, and 2.4 in frontal cortices. In DS adults, the levels of DYRK1A protein in the temporal, occipital
cortex and corpus callosum were higher than in the controls by a factor ranging from 1.4 to 2.4 (p< 0.05).
These results support “gene dosage” mechanism of DYRK1A protein in pathology seen in people with DS.
2. DYRK1A protein isoforms 1 and 2. Alternative splicing of exon 6 produces mRNAs coding two major
isoforms of DYRK1A protein: isoform 1 (763 aa) and isoform 2 (754 aa). In human and mouse brain
extracts, isoform1-specific antibody recognized three bands (97-94 kDa doublet and 87 kDa singlet),
whereas isoform2-specific antibody detected exclusively 97-94 kDa doublet. The highest levels of both
isoforms were observed in fetuses and few-day-old mice, and the lowest in 2-month and 6-month-old
mice, in which the level of isoform 1 was reduced by 65% and isoform 2 by 80% compared to fetuses.
Significantly faster reduction of isoform1 than 2 in first two weeks of life suggests difference in the role of
isoforms 1 and 2 in developing brain.
3. Contribution of DYRK1A to neurofibrillary degeneration. Recombinant DYRK1A phosphorylates
recombinant tau to a stoichiometry of ~1.2 mol phosphates/mol of tau. DYRK1A phosphorylates tau at
several sites with an efficiency order of Ser212>Ser199/Ser396>Ser202/Thr205/Ser404. In DYRK1A
transfected PC12 cells tau is hyperphosphorylated at the Ser199 and Thr212. Te examination of the level
of tau phosphorylation in brain tissue from 6 DS (~60 years old) and 6 age-matched controls by Western
blots revealed that: (1) over-expression on DYRK1A results in increased kinase activity in DS brains, (2)
DYRK1A phosphorylates tau at several phosphorylation sites to a physiologically relevant level in vitro and
in cultured cells, (3) these sites of tau are hyperphosphorylated in brains of adults with DS, (4)
phosphorylation of tau by Dyrk1A primes tau for phosphorylation by GSK3beta at several phosphorylation
sites. Immunocytochemistry of brain tissue showed that DYRK1A accumulates in neurofibrillary tangles
(NFTs) in people with sporadic AD and in subjects with DS/AD but not in control subjects. Overexpression
of DYRK1A in people with DS was associated with the presence of DYRK1A-immunoreactivity in 50% of
NFTs whereas in sporadic AD less than 10% of tangles were DYRK1A-positive. These data indicate that
neurofibrillary degeneration in DS is modified by DYRK1A in gene dosage-dependent manner.
54
Po s t e r S e s s i o n 1
P O S T E R
S E S S I O N
1
WEDNESDAY, MARCH 7, 2007
6:30 P.M. - 8:30 P.M.
55
1.
POSTER SESSION 1
Hyporesponsive Sensory Patterns in Young Children with Autism,
Developmental Delay, and Typical Development
Grace T. Baranek, Margaret DeRamus, Linda R. Watson,
Brian A. Boyd & Michele D. Poe
Correspondence to: Grace Baranek, CB# 7122, 2050 Bondurant Hall,
University of North Carolina at Chapel Hill, Chapel Hill, NC 27599-7122
[email protected]
Introduction: The exploration of sensory features in autism is pertinent due to the prevalence of such
patterns. These features are widely described by professionals, families, and persons with autism,
although they are not included in diagnostic criteria. The objective of this study was to determine the
specificity and developmental correlates of a hyporesponsive sensory pattern in a sample of young
children with autism and related disorders. Hyporesponsiveness refers to lack of response, or insufficient
intensity of response to sensory stimuli. The focal question is whether or not children with autism show a
generalized pattern of hyporesponsiveness and to what degree this pattern is affected by contextual
factors (social vs. nonsocial), stimulus modalities (tactile vs. auditory vs. visual), and/or maturational
variables. We hypothesized that children with autism would differ significantly in level of
hyporesponsiveness from comparisons (i.e. developmental delay, typical), even when controlling for age.
Method: The hyporesponsive scale from the Sensory Processing Assessment for Young Children (SPA)
(Baranek, 1999) was used to identify latency to orient to sensory stimuli. Six items (name call, hand wave,
shoulder tap, sound, light, and air puff), counterbalanced for context (social/nonsocial) and modality
(tactile/ visual/auditory) were administered to 58 children with autism (MA 25.1, sd 18.1), 59 children with
typical development (MA 30.1, sd 20.32), and 34 children with developmental delays (DD) (MA 26.0, sd
14.1). Within the group of DD, 14 children had Down Syndrome. Interrater reliability with >90% agreement
was established by scoring and analyzing data between two raters. Additional diagnostic (e.g. ADOS) and
developmental (e.g. Mullen, Vineland) assessments were administered to confirm each child’s group
assignment and to describe sample characteristics. For analysis, a repeated measures mixed model with
social and non-social scores as the outcome was run, controlling for the mother’s education and gender,
and another model was run with modality scores. In keeping with the conference theme, we will also
analyze a Down Syndrome subgroup to explore syndrome-specific characteristics.
Results: There were significant differences between the groups in the relationship between mental age
and hyporesponsiveness. Autism subjects had a greater decline (steeper slope) than typical and DD
subjects due to higher levels of hyporesponsive patterns at lower mental ages. The autism group also
displayed more difficulty with orienting than the controls; the social context exceeded group differences
found in the nonsocial context. There were also significant group differences in orienting responses based
on modality.
Discussion: Results indicated that impaired sensory orienting may be a feature that is more specific to
children with autism versus comparisons. However, MA explains much of variance associated with
responsiveness to stimuli across groups, so as children advance cognitively, responsiveness improves.
Although group differences were found, the context of sensory stimuli (social or non-social) affected the
orienting response for all children, indicated by the finding that social stimuli were more difficult across
groups, particularly at lower MAs. Theoretical implications and limitations (e.g., salience of social versus
nonsocial stimuli) will be discussed. In addition, results from analysis of a subgroup of children with Down
Syndrome will be presented. Future studies would benefit by using similar analyses that control for effects
of MA and CA through the use of mixed models rather than traditional matched group designs.
Acknowledgements: Grant NICHD R01-HD-42168
56
2.
POSTER SESSION 1
Intrauterine Growth Retardation and Executive Function Performance at School Age
Richard C. Belser, Vicki Sudhalter, Judith M. Gardner & Bernard Z. Karmel
N.Y.S. Institute for Basic Research in Developmental Disabilities
1050 Forest Hill Road, Staten Island, New York 10314
Contact: [email protected]
Introduction: Continuing advances in prenatal and postnatal care have led to an increased survival rate
of infants considered to be at risk for developmental impairment from various causes, including chronic
intrauterine stress during pregnancy. A common outcome of such prenatal stress is intrauterine growth
retardation (IUGR). Although related to prematurity and low birth weight, IUGR refers specifically to
infants who are significantly smaller than would be expected for their gestational age. IUGR has
sometimes been associated with poor neurodevelopmental outcome, although the generality of such a
relationship has been questioned (Burke et al., 1990). The data being presented here address the
relationship between the degree of IUGR in infants with no evident structural CNS injury, and the
subsequent development of executive function skills at school age.
Methods: The Wilding Monster Sorting Task (WMST), a child-friendly version of the Wisconsin Card
Sorting Task, was administered to children between five and six years of age as part of a larger ongoing
longitudinal nerurodevelopmental study. The WMST is a computerized task that requires participants to
assign monsters to one of four teams, each led by a monster king pictured across the top of the monitor.
Each king exemplifies a different combination of values of three features (shape of face, number of eyes
and color of feelers). As new monsters appear at the bottom of the screen, the participants must assign
them to a team by choosing a king, who then either smiles or frowns to indicate response correctness.
After reliably learning to base team selection on a certain feature, the feature rule is changed without
notice. Performance is measured by the number of trials on which a correct response is made. Our
measure of IUGR is based upon Fenton’s (2003) guidelines for the growth assessment of preterm babies.
Results: The data from a preliminary sample of 32 children were examined by multiple regression
analysis (using Statistica) to determine the influence of IUGR and age upon WMST performance. The
contribution of IUGR to outcome was significant [beta = 0.42, p=.02]. The contribution of age was not
significant.
Discussion: Good performance on the WMCS requires both the ability to learn conceptual rules, and the
ability to inhibit responding according to a previously correct rule after that rule has changed. Although our
preliminary sample is not adequate to assess the relative contribution of these two factors to the children’s
performance, they both represent executive function skills that emerge during school age and may be
susceptible to early developmental problems associated with IUGR. As additional data are collected and
analyzed, we expect to be able to provide a more comprehensive description of the relationship between
IUGR and executive function development.
Key References:
Burke, G., et al. (1990). Is intrauterine growth retardation with normal umbilical artery blood flow a benign
condition? British Journal of Medicine, 300, 1044-1045.
Fenton, T. R. (2003) A new growth chart for preterm babies: Babson and Benda’s chart updated with
recent data and a new format. BMC Pediatrics. 3, 13-22.
57
3.
POSTER SESSION 1
Persistence in Early Communication by Young Children with Down Syndrome and
Other Developmental Disabilities
Nancy C. Brady and Kandace Fleming
University of Kansas
Contact:[email protected]
Introduction: During early stages of communication development, children experience frequent
communication breakdowns. That is, their communication partners often ask for clarification, change topic,
or ignore children’s attempts to communicate. The ability to negotiate these episodes of communication
breakdown enables children to continue the conversational exchange and eventually communicate their
desires and interests to their partners. Children with Down syndrome are often significantly delayed in
learning to talk and when they do begin speaking they may be difficult to understand (Roberts et al.,
2005). Therefore, children with Down syndrome may experience even more frequent communication
breakdowns than other children in similar developmental stages. In addition, young children with Down
syndrome have been described as showing a lack of persistence compared to typically developing
children and may be less likely to persist in their communication attempts when they are misunderstood,
The purpose of this study was to compare episodes of communication breakdown during mother-child
interactions across children with Down syndrome, children with developmental disabilities associated with
other etiologies, and typically developing children matched for language development.
Methods: Eight children between the ages of 38 and 52 months (mean 44.6) with a diagnosis of Down
syndrome were individually matched to eight children who had developmental disabilities but not Down
syndrome (mean CA 40 mo.), and eight typically developing toddlers (mean CA 19 mo.), on the basis of
receptive language age and expressive language level. All of the children were in early stages of
expressive communication development, producing less than 10 different words through speech or sign.
Some children did not yet produce any words.
Children participated in 30 minutes of mother-child interaction that was videotaped through a one-way
mirror. The videotapes were later coded for child initiations, maternal responses to these initiations and
whether or not the child succeeded in conveying their apparent intent. Child communication acts were
described in terms of communication forms (gesture types, vocalizations, etc.) and communicative
functions (e.g., behavior regulation, joint attention).
Results: Children with Down syndrome initiated more communication acts than the other two groups. All
children experienced similar proportions of communication breakdowns during mother-child interactions. In
addition, children with Down syndrome were significantly more successful in resolving communication
breakdowns than children in the other groups. Children were more likely to initiate and repair joint attention
acts, but there was not a significant difference across groups.
Discussion: Children with Down syndrome were persistent in their communication attempts and were
more successful at repairing communication breakdowns than other children who were at similar language
levels. It may be that lack of persistence occurs later in development or does not apply to communication
breakdowns. These results should be interpreted with caution, however, due to the small number of
participants in each group.
58
4.
POSTER SESSION 1
Experiences of Parental Advocates in Self-Help Groups for Children with Autism
Irene Carter
University of Calgary
877 Westchester Drive
Windsor, Ontario
N8S 3Y6
Contact: [email protected]
This qualitative study explored the experiences of parental advocates in attending self-help groups for
children with autism. Twenty-two interviews with participants indicated the limitations of current social
policies related to chronic disabilities, gender issues, and service delivery and the need to seek
information and interventions through participation in self-help groups. The positive experiences included
knowledge of interventions, rights and legislation, increased self-esteem, empowerment, hope, an
appreciation of the care-giving role, and increased creative and flexible self-help group strategies. The
negative experiences included increased stress associated with decreased time for family and work, and
frustrations and disappointments in outcomes.
Participants found self-help groups as necessary, and as serving a role in protecting vulnerable. While
appreciative of the enabling group support, education and advocacy provided in self-help groups, study
participants also noted a certain amount of ambivalence about membership and indicated group practices
in need of improvement. The findings supported existing literature and contributed to the nascent literature
on negative experiences.
Suggestions for improved group practices included the use of professional assistance, improved feedback
mechanisms, and the development and maintenance of a clear group focus. Implications for social work
suggest the importance of professional training on how best to best assist self-help groups in developing
strategic alliances and collaborative community initiatives. The study provided opportunities for parental
advocates in self-help groups, professionals, and the public to better understand the challenges facing
those who are inspired to provide positive change for children with autism.
59
5.
POSTER SESSION 1
Early Learning Profile in Preschoolers with Williams Syndrome: A Descriptive Study
Ashley Cole, David Most, Deborah Fidler,
Amy Philofsky, & Susan Hepbun
Colorado State University; University of Colorado Denver Health Sciences Center
Contact: [email protected]; HDFS Dept. 502 W. Lake St. Fort Collins, CO 80526
Williams syndrome is a neurodevelopmental, multisystem disorder resulting from an abnormality on
chromosome 7 (Ewart et al., 1993). Mervis, Klein-Tasman, and Mastin (2001) examined the adaptive
behavior of 4-through 8-year-olds with Williams syndrome and identified patterns of relative strengths and
weaknesses in this population. This study and others have established that children with Williams
syndrome typically have relative strengths in the areas of expressive language, emotional responsivity and
social interest and relative weaknesses in the areas of receptive language and fine motor skills, but much
remains to be learned about how these splits in ability emerge and develop in the earliest years of life
(Hepburn et al., 2005).
This study aims to contribute to the literature characterizing the early manifestation of this phenotypic
profile in young children with Williams syndrome and to describe the magnitude of the dissociation
between their relative strengths and weaknesses. The Mullen Scales of Early Learning (MSEL) was
administered to a chronological age and developmentally matched group of 18 children with Williams
syndrome (age M = 35.75 months, SD = 7.79) and 17 children with developmental disabilities of mixed
etiologies (age M = 36.65 months, SD = 7.85). Groups were similar on overall MSEL standard score (WS
M = 61.94, SD = 10.00; DD M = 61.35, SD = 61.35). Four subscales were administered: fine motor,
expressive language, receptive language, and visual reception.
Results showed that children with Williams syndrome demonstrated a different early learning profile than
children in the developmental disabilities comparison group. Specifically, children with Williams syndrome
showed an overall advantage for verbal versus nonverbal functioning, while children in the comparison
group showed a slight advantage for nonverbal functioning. Children with Williams syndrome showed a 9
month advantage over the comparison group children in the domain of expressive language (WS M =
27.22 months, SD = 9.83; DD M = 18.41, SD = 6.81), and a 4 month advantage over the comparison
group children in the receptive language domain (WS M = 24.88, SD = 8.50; DD M = 20.88, SD = 9.78).
In contrast, the comparison group children showed a 3 month advantage over the Williams syndrome
group in the fine motor domain (WS M = 19.88, SD = 4.52; DD M = 22.94, SD = 7.20).
In addition, children with Williams syndrome on average showed a 2.3 month (SD = 5.69) advantage for
expressive language functioning over receptive language functioning, while children in the mixed
comparison group had an average advantage of 2.47 (SD = 7.22) months for receptive language over
expressive language. A 95% confidence interval for this between group difference is (.34, 9.26). While
children with Williams syndrome showed similar performances on the visual receptive and receptive
language domains of the MSEL (a .83 month advantage for receptive language skills), children in the
mixed comparison group showed a 3.41 month advantage for visual receptive over receptive language
performance. A 95% confidence interval for this between group difference is (7.02, 7.78). Similarly,
children with Williams syndrome on average showed a 3.16 month advantage for expressive language
over visual receptive skills, while children in the comparison group showed a 5.88 month advantage for
visual receptive over expressive language skills. A 95% confidence interval for this between group
difference is (4.73, 13.36). Implications for early intervention planning will be discussed.
60
6.
POSTER SESSION 1
Predicting Marital Satisfaction from Initial Maternal Depression and Burden of Care
Jenalee Coster, Brian M. Jobe, and Laraine M. Glidden
St. Mary’s College of Maryland
Campus Center 1410, 16800 Point Lookout Rd, St. Mary’s City, MD 20686
Contact: [email protected] and [email protected]
Introduction: The presence of a child with developmental disabilities (DD) presents unique care giving
requirements that may strain the marital relationship. In a meta-analysis Risdal and Singer (2004)
examined the occurrence of divorce and marital discord in married couples rearing a child with DD in
comparison with controls. They found a significant effect in the predicted direction, but it was small.
Nonetheless, it is still important to determine the risk factors related to rearing a child with DD that predict
marital tension and disruption. In the current study we examined the effect of the mother’s initial
depression and burden of care, as measured by available resources and perceived child demands, on
marital satisfaction. We hypothesized that reports of greater initial depression along with a heavy burden
of care would predict more negative reports of marital satisfaction. In addition to main effects for these
two variables, we also hypothesized that caretaker burden would interact with depression such that
mothers with low levels of depression would have high marital satisfaction regardless of their burden of
care.
Methods: The sample is a subset of 53 mothers and their spouses from a sample of 249 families that
participated in a 19-year longitudinal study of families raising children with DD. Data were collected for
four different time periods, but the current study examines data only from Time 1, Time 2, and Time 3.
Maternal depression was measured by the Beck Depression Inventory (BDI) at Time 1. Burden of care
was measured by the Friedrich Questionnaire on Resources and Stress (QRS) at Time 2, and marital
satisfaction was measured using the Locke-Wallace Marital Adjustment Inventory at Time 3.
Results: To test the moderated model, BDI at Time 1 and the QRS at Time 2 were converted to z-scores
and an interaction term computed. Using a multiple regression, the mother’s BDI scores, her QRS scores,
and the interaction were entered as predictors of marital satisfaction for both mothers and fathers at Time
3. The hypothesis of a moderated model was not supported. Initial maternal depression and burden of
care were unique predictors of maternal marital satisfaction. Maternal burden of care was the sole
predictor of father marital satisfaction.
Discussion: In sum, maternal burden of care was a significant predictor of marital satisfaction for both
mothers and fathers. Initial maternal depression, however, contributed to marital satisfaction scores only
for mothers. The two variables of caretaker burden and initial maternal depression are highly correlated,
but they do not interact to create a moderated effect on future marital satisfaction. It is possible that
mothers with higher depression receive more help from their husbands in caring for the children and that
this sharing of responsibilities of care may increase scores on the Locke-Wallace, thus offsetting the
predicted moderating effect in the opposite direction.
References:
Glidden, L. M. & Schoolcraft, S. (2003). Depression: its trajectory and correlates in mothers
rearing children with intellectual disability. Journal of Intellectual Disability Research. 47, 250-263
Risdal, D. & Singer, G. H. S. (2004). Marital adjustment in parents of children with disabilities:
A historical review and meta-analysis. Research and Practice for Persons with Severe Disabilities,
29, 95-103.
61
7.
POSTER SESSION 1
Factors Predicting Mortality in a Residing with Family Aging Sample
of Adults with Mental Retardation
Anna J. Esbensen, Marsha Mailick Seltzer, & Jan S. Greenberg
Waisman Center, University of Wisconsin – Madison
Contact: Anna J. Esbensen, Waisman Center, room 561,
1500 Highland Avenue, Madison, WI, 53705, [email protected]
Introduction: Studies on the mortality and morbidity of adults with Down syndrome relative to their age
peers with other types of mental retardation report a pattern of similar rates of mortality until age 40, but
elevated mortality rates thereafter. However, the life course of adults with Down syndrome has been
extending dramatically. We investigated whether the prior findings would replicate in an aging sample of
adults with Down syndrome or other types of mental retardation who were co-residing with their families.
Specifically, we ask whether there were differences in the pattern of mortality between the two groups,
whether there were differences in the causes of death, and if we could determine predictors of mortality.
Method: Interviews were conducted with 461 mothers over the age of 55 caring for a co-residing adult son
or daughter with mental retardation. Mothers were interviewed at 18 month intervals from 1988 to 2000
and provided information about their son or daughter’s health, behavior problems and functional abilities
(Krauss & Seltzer, 1999). The mean age of the child was 33 years at the start of the study. Of the original
sample, 169 children had Down syndrome and 291 had other types of mental retardation. Mortality, age at
death and causes of death during the course of the study were obtained from the mother. For deaths
occurring after the study was completed, the National Death Index (NDI) is the source of data regarding
mortality, age at death and the cause of death.
Results: Of the original sample, 31 individuals with Down syndrome and 42 individuals with other types of
mental retardation died. There were no significant differences between Down and comparison cases in the
proportion of deaths, P2(1)=1.22, p = .17, nor the age of death, t(71)=1.51, p=.13. Differences between
Down and comparison cases in the number of deaths caused by Alzheimer’s, heart problems, unknown or
other causes will be reported. Predictors of mortality were entered into a logistic regression as follows: (1)
Down syndrome, gender, level of mental retardation, (2) initial status of health, behavior problems and
functional abilities, (3) change in health, behavior problems and functional abilities, and (4) age. In the final
model, age (Exp$=1.12, p=.00), initial level of functional abilities (Exp$=.42, p=.02), change in functional
abilities (Exp$=.00, p=.00), change in behavior problems (Exp$=2.26, p=.01), and a diagnosis of Down
syndrome (Exp$=2.46, p=.04) were significant predictors of mortality of the adult. The results remained
consistent when indicators of epilepsy, placement, and parental death were added into the model.
Discussion: Having a diagnosis of Down syndrome did not impact the number of deaths, or the age at
death in this co-residing sample of adults with mental retardation. However, when other contextual factors
were considered, such as initial age and decline in functional abilities, Down syndrome was found to be a
significant predictor of mortality. The current findings indicate that a diagnosis of Down syndrome is
predictive of mortality when individual differences in functional decline are taken into account.
Key References:
Krauss, M. W., & Seltzer, M. M. (1999). An unanticipated life: The impact of lifelong caregiving. In H.
Bersani (Ed.), Responding to the challenge: International trends and current issues in
developmental disabilities. Brookline, MA: Brookline Books.
62
8.
POSTER SESSION 1
Examining Differences between Autistic Children with and without Mental Retardation (MR)
Marygrace Y. Kaiser, Vanessa Gonzalez, & Jennifer S. Durocher,
Michael Alessandri, ([email protected]) and Claudia Rojas, ([email protected])
University of Miami Flipse Building P.O. Box 248185 Coral Gables, FL 33124-0751
[email protected], [email protected], & [email protected] ,
It is widely accepted that mental retardation is a feature which is frequently associated with Autism
Spectrum Disorders (ASDs). In fact, research has consistently indicated a high degree of comorbidity
between autism and mental retardation, with estimates commonly cited rates of upward of 70 to 75%
(DSM-IV, 1994; Ozonoff & Rogers, 2003; Siegel, 1997). Better characterization of these two subgroups is
needed, in order to understand how these groups differ with respect to factors such as symptom
presentation and age of diagnosis, educational eligibilities and placement, and follow-up cognitive
evaluations, as these factors are likely to affect long-term outcome. In an attempt to address these issues,
this study will utilize a unique community sample of children with Autistic Disorder to examine (1) types of
tests used to asses IQ, (2) symptom presentation via the Social Communication Questionnaire (SCQ), (3)
ages at which children were assessed by professionals, and (4) school eligibility and placement.
This study consisted of 77 children with a parent reported diagnosis of Autistic Disorder, mostly consisting
of boys as is common with this disorder. The parents of these children participating in a larger ongoing
prevalence study funded by the CDC developed to monitor and track the prevalence of ASDs in MiamiDade County. Of these children, 39 had an IQ score less than 70 and the remaining 38 had IQ scores of
70 or above. IQ results were obtained via historical records obtained from each child at varying ages.
A wide range in the type of test used to assess IQ was found in this sample. It was also found that the
“high IQ” group were significantly more likely to have received a test that emphasized nonverbal IQ. There
was also a significant mean difference in the age at which children were first seen by a professional for
parental concern as well as when they were administered their first IQ evaluation. Children in the “low IQ”
group were younger when first seen by a professional as well as at their first evaluation than children in
the “high IQ” group. It was also observed that children in the “high IQ” group were significantly less likely
to have an Autism eligibility and were significantly more likely to be placed in general education
classrooms than were the “lower IQ” group. No significant difference was found for total SCQ score
between the means for the two groups. However, risk ratios indicate that children in the “lower IQ” group
were more likely to score above the threshold for Autism vs. ASD on the SCQ. The vast majority of
children in this sample did not receive any follow-up IQ testing after their initial evaluations, therefore
analyzing change in IQ was not possible.
It may be that the higher IQ’s obtained for this group may be a function of the type of test used; in other
words, perhaps the “lower IQ” group might have obtained higher IQ estimates had they been given a
nonverbal test. It was also found that lower IQ children were seen by professional and evaluated earlier
indicating that for children exhibiting comorbidity, their deficits were more obvious to parents which caused
concern earlier on than for children with higher IQ’s. The finding that the “high IQ” children were less likely
to have an Autism label may also be related symptom presentation. It may be that school IEP teams
perceive children who are more cognitively capable and less affected behaviorally to be more likely to
benefit from a general education environment.
Ozonoff, S. and Rogers, S. J. (2003). From Kanner to the millennium: scientific advances that have shaped clinical
practice. In S. Ozonoff, S. J. Rogers, & R. L. Hendren (Eds.), Autism Spectrum Disorders: A Research Review
for Practitioners. (pp. 3-33). Virginia: American Psychiatric Publishing Inc.
Siegel, B. (1996). Getting a diagnosis. In B. Siegel (Ed.), The World of the Autistic Child: Understanding and Treating
Autistic Spectrum Disorders. New York: Oxford University Press.
63
9.
POSTER SESSION 1
Linking Executive Function and Repetitive Behaviors in Autism Spectrum Disorders
Harrison, B.1, Black, D.2, Wallace, G. 3, Della Rosa, A.1, & Kenworthy, L.1
1
Children’s National Medical Center, Washington, D.C., 2Boston University School of Medicine,
and the 3National Institute of Mental Health
Contact: Bryan Harrison, Center for Autism Spectrum Disorders,
14801 Physician’s Lane, Suite 173, Rockville, MD 20850 ([email protected]).
Introduction: Identification of autism spectrum disorder as a multi-gene disorder drives interest in
ascertaining the neuropsychological correlates associated each of the triad of behavior clusters associated
with autism. Executive function (EF) is an obvious candidate to consider when investigating the cognitive
phenotype underlying the repetitive behaviors cluster of autism symptoms. Previous research (Lopez,
Lincoln, Ozonoff, & Lai, 2005) supports a relationship between executive function profiles and repetitive
behaviors. Other investigations explored the relationship among the many discrete behaviors associated
with the diagnosis of autism through factor analysis of parent responses to gold standard diagnostic
interviews. Cuccaro (2003) and Szatmari (2006) each posited a two-factor model of repetitive and
stereotyped behavior: one domain comprised of sensory/motor behaviors and another encompassing
pervasive cognitive rigidity (or insistence on sameness). The present study examines the factor structure
of repetitive/stereotyped behavior items from the ADI-R in a sample of high-functioning children with
autism spectrum disorders, and the relationship of the factor structure to cognitive measure of executive
functioning.
Methods: Participants: 114 high functioning children (n=94 [83%] male; mean age = 8.13, range = 2-17)
diagnosed with an autism spectrum disorder based on ADI-R, and expert clinical impression. All
participants received a clinical neuropsychological evaluation, and their parents were administered the
ADI-R (Lord, Rutter, & Le Couteur, 1994). An exploratory principal components factor analysis with
Varimax rotation was completed using algorithm items assessing repetitive/stereotyped behavior from the
ADI-R. Questions about verbal rituals and stereotyped speech were included in the factor analysis, with
the a priori assumption that scores on these two items reflect repetitive/stereotyped behavior. Factors
were then correlated with IQ and select measures of executive functioning assessed by the Test of
Everyday Attention in Children (Tea-Ch).
Results: The factor analysis yielded a 4-factor solution (eigen values > 1.0) accounting for 63% of the
total variance. Inspection of items that comprised each factor suggested the following constructs: Factor
1: ritualistic behavior; Factor 2: circumscribed interests and repetitive behavior; Factor 3: preoccupation
with discrete perceptual elements; and, Factor 4: stereotyped speech. Factor 1 was not correlated with
any of the selected neuropsychological measures; Factor 2 was negatively correlated with verbal IQ (.232, p < .05); Factor 3 was negatively correlated with organized visual search ability (-.510, p < .05) and
inhibition (-.435, p < .01); Factor 4 was negatively correlated with organized visual search ability (-.522, p <
.01).
Conclusion: Results of the factor analysis yielded a 4-factor solution in our high functioning ASD sample
in contrast with earlier reports of a two factor structure (Cuccaro, et al., 2003; Szatmari, et al., 2006). We
found a specific factor relating to preoccupation with discrete perceptual elements (Factor 3); this factor
negatively correlated with a task tapping organized visual search strategies, as well as a measure of
inhibition. This relationship may reflect an underlying inability to integrate information in autism spectrum
disorders. Our results support the promise of attempts to link the diagnostic behaviors of autism to
underlying cognitive constructs (or intermediate phenotypes) and suggest that specific executive functions
may serve as empirically valid links from behavioral symptoms to brain function.
References:
Lopez, B.R., Lincoln, A.J., Ozonoff, S., & Lai, Z. (2005). Examining the relationship between executive
functions and restricted, repetitive symptoms of autistic disorder. Journal of Autism and
Developmental Disorders, 35(4), 445-460.
64
10.
POSTER SESSION 1
Restricted, Repetitive and Stereotyped Behaviors, Interests,
and Activities in Children with Autism
Athena Hayes, University of Wyoming, [email protected]
William MacLean, Jr., University of Wyoming, [email protected]
Susan Hepburn, University of Colorado Health Sciences Center, [email protected]
Sally Rogers, University of California-Davis, [email protected]
Introduction: Although restricted, repetitive behaviors and interests are one of the three core defining
features of autism, the extent to which these behaviors are specific to autism is not clear, leading some to
propose that these behaviors may not be a necessary diagnostic requirement. Comparing restricted,
repetitive behaviors and interests in autism with behaviors exhibited by other diagnostic groups could allow
for further delineation of the autism behavioral phenotype.
Methods: Children were administered an intensive diagnostic battery that included the ADOS-G and the
ADI-R. Differences in the severity and patterns of repetitive behavior were examined among three wellmatched, well-defined diagnostic groups {autism (n = 25), fragile X syndrome (n = 10), and
developmentally delayed (n = 25)}, through discriminant function analysis.
Results: Repetitive behavior was apparent across all three diagnostic groups; yet, there were observable
differences in the severity and pattern of behavior displayed allowing some differentiation between groups.
Children with autism displayed significantly more unusual sensory interests than the fragile X syndrome
and developmentally delayed groups and more unusual preoccupations than the developmentally delayed
group. Further, children with autism displayed a pattern of high unusual sensory interests and relatively
low levels of hand and finger mannerisms and self-injurious behavior as compared to the other groups.
Discussion: Although there does not appear to be a repetitive behavior specific to autism based on these
results, the occurrence, severity, and pattern of repetitive behavior exhibited provides support for the
inclusion of repetitive behavior as a diagnostic criterion for autism. Further examination of the various
forms of repetitive behavior is critical in charting the diagnostic stability of autism. Examining all aspects of
the disorder in a systematic fashion allows for a further delineation of the broader autism phenotype.
References:
Bodfish, J. Symons, F. J., Parker, D.E., Lewis, M.H. (2000). Varieties of repetitive behavior in autism:
Comparisons to mental retardation. Journal of Autism and Developmental Disorders, 30(3), 237243.
Lord, C. (1995). Follow-up of two-year-olds referred for possible autism. Journal of Child Psychology
and Psychiatry, 36(8), 1365-1382.
Turner, M. (1999). Annotation: Repetitive behavior in autism: A review of the psychological research.
Journal of Child Psychology and Psychiatry, 40(6), 839-849.
65
11.
POSTER SESSION 1
Enhancing Cognitive-Emotional Skills for Children with Developmental Disabilities
through Computer-Based Training
Ingrid M. Hopkins, Margaret J. Bates, Shana B. Crowson,
Rebekah K. Pearson, and Fred J. Biasini
University of Alabama at Birmingham
CH19, Suite 307, 1530 3rd Avenue South, Birmingham, AL, 35294-2041
Contact: [email protected]
Previous research has highlighted deficits in social relations and non-verbal communication in both
children with Autism and Mental Retardation. Specifically, earlier studies have shown that children with
Autism and Mental Retardation perform significantly worse on tasks involving emotion recognition as
compared to typical children. Also, individuals with Autism and Mental Retardation show marked
impairments in the use of multiple nonverbal behaviors such as facial expressions and gestures to
regulate social interactions. The overall purpose of the current study was to use recent advances in
research, theory, and technology to develop, implement, and evaluate an avatar tutor for social skills
training in children with Autism or Mental Retardation. Avatars are computer embodied virtual people that
have a knowledge base and the ability to converse with humans in natural language. The advantages of
employing computer-controlled applications for training include the unique ability to control and manipulate
the visual and auditory components of spoken language automatically, enough variation in the learning
environment to facilitate generalization of what is learned, and a one-on-one format between the child and
the computer. “Face Say” was developed as a colorful program that contains several different activities
designed to teach children specific social skills, such as eye gaze, joint attention, and facial recognition.
The interactive features of the software provide opportunities for children to respond to social situations.
The tasks differ in difficulty to assure that participants of various levels can be successful as well as
challenged by the tasks. The children were asked to attend to the games for six weeks. Preliminary
results indicate that the parents of the children with Autism and Asperger Disorder reported an
improvement in their children’ social skills (e.g. cooperation, responsibility, self control) after participating in
the study, t(7) = -3.257, p = 0.014. Second, there was a decrease in overall reported problem behaviors,
t(7) = 3.112, p = 0.017 and internalizing behaviors, t(7) = 3.819, p = 0.007. Finally, the children showed
improvements in their levels of cooperation, t(7) = -5.292, p = 0.001. The findings of this study are of
great importance. It provides information about the benefits of computer-based training for children with a
variety of developmental disabilities, including Autism, Asperger Disorder, and Mental Retardation. The
knowledge gained from this study could be used to create more effective and cost efficient therapy for
improving the cognitive-emotional skills of children with developmental disabilities. Future research
directions are discussed.
References:
Bernard-Opitz, V., Sriram, N., & Nakhoda-Sapuan, S. (2001). Enhancing social problem solving in
children with autism and normal children through computer-assisted instruction. Journal of Autism
and Developmental Disorders, 31, 377-398.
Bosseler, A., & Massaro, D.W. (2003) Development and evaluation of a computer-animated tutor for
vocabulary and language learning in children with Autism. Journal of Autism & Developmental
Disorders, 33, 653, 673.
Yirmiya, N., Erel, O., Shaked, M., & Solomonica-Levi, D. (1998). Meta-analyses comparing theory of mind
abilities of individuals with Autism, individuals with Mental Retardation, and normally developing
individuals. Psychological Bulletin 124(3), 283-307.
66
12.
POSTER SESSION 1
Increased Special Health Care Needs in Children with Severe Developmental Disabilities
and Behavioral Disorders: Implications for Interdisciplinary
Treatment of Problem Behavior
Craig H. Kennedy, A. Pablo Juárez, Angela Becker, Kathryn Greenslade,
Mark T. Harvey, Clare Sullivan, and Brenna Tally
Vanderbilt University
Objective: Behavioral disorders among children with severe developmental disabilities (CSDD) have a
negative impact on life quality. Several single-case design studies have suggested that special health
care needs (SHCNs) may contribute to the prevalence and intensity of behavioral disorders in this
population. However, no direct comparison has been conducted to assess a possible association between
the health status of CSDD and the occurrence of behavioral disorders. Methods. We used a matchedcomparison control group design to establish whether SHCNs were higher in CSDD with behavioral
disorders versus CSDD without behavioral disorders. Children were matched on age, gender, race,
mental retardation level, and DSM-IV axis I diagnosis (N=36). Measures included the Child Behavior
Checklist, direct behavioral observation, health status exam, and Childhood Health Questionnaire (CHQ).
Results. CSDD with behavioral disorders had significantly higher levels of SHCNs as measured by the
CHQ and health status exam. Overall, CSDD with behavioral disorders had a three-fold higher incidence
of SHCNs than CSDD without behavioral disorders. No between group differences were observed in the
number or types of prescription medications children received. Conclusions. CSDD with behavioral
disorders have a higher incidence of SHCNs than CSDD without behavioral disorders. It is possible that
SHCNs contribute to the occurrence and/or intensity of behavioral disorders in his population. Our
findings suggest that the treatment of behavioral disorders in CSDD requires interdisciplinary care
coordination among health, educational, and behavioral professionals if SHCNs are present.
67
13.
POSTER SESSION 1
Teaching Money Skills to Children with Intellectual Disabilities: Rapid Learning via
Constructed Response Matching-to-Sample Procedures
Joanne B. Kledaras & Dana Hurlbut, Praxis, Inc.271 Waverley Oaks Road, Suite 206 Waltham, MA 02452
William J. McIlvane & William V. Dube, UMMS Shriver Center, 200 Trapelo Road, Waltham, MA 02452
[email protected]
This project is developing and evaluating state-of-the-art computer-based methods for teaching money
skills prerequisites to persons with autism, mental retardation, and other intellectual disabilities. The
foundation for the project is a long-term program of basic and translational research supported by NICHD.
That research documented exceptional potential of stimulus equivalence and related stimulus control
shaping techniques for teaching persons who would not ordinarily be expected to master money skills.
These achievements, however, depended upon a highly intricate set of procedures – the complexity of
which rendered the approach unsuitable for routine implementation in special education settings.
The research dimension of the current project is three-fold: First, it is evaluating an enhanced version of
the money skills teaching program that replaces human decision-making with algorithmic specification of
necessary teaching steps and sequences. Second, it is evaluating new algorithms for proscriptive
programming and error-handling. Third, the project is evaluating the human interface that supports the
efforts of teachers to use the program effectively.
The project is also re-evaluating the translational research on which the program is based. One
achievement has been an elegant solution to an ongoing problem – teaching children to indicate “no more”
when none of the coins available in a “choice pool” can be combined to make a valid match in the
constructed-response matching format. In prior research, the child was required to respond to another
location to indicate “no more” – behavior that proved difficult to establish in a subset of children. The
solution was to place a token in the pool. The child could respond to it to indicate “no more;” inhibiting
responses to coins in the pool was no longer required.
Other findings – quite unexpected – were data collected in an effort to refine pre-intervention
assessments. During assessment, we observed apparent learning of new performances despite the
absence of explicit teaching. The table below presents illustrative data from a subset of children – each
child’s initial accuracy at each of five coin-equivalence values (5¢, 10¢, 15¢, 20¢, and 25¢), his/her highest
score achieved during the assessment, and the number of sessions preceding his/her highest score. That
children achieved non-zero initial scores may be due to the fact that they had coin skills as IEP objectives
and were thus beneficiaries of prior teaching by others (only partially successful as the frequently low
initial scores also show).
Value
5¢
10¢
15¢
20¢
25¢
Student First
High
Sess
First
High
Sess
First
High
Sess
First
High
Sess
First
High
Sess
S3
73
100
7
61
87
4
61
88
17
48
86
7
50
80
11
S8
74
100
9
66
100
10
50
100
14
29
100
14
45
85
12
S16
87
100
5
78
96
9
46
79
9
56
81
13
63
81
12
We attribute increases shown by many children during our assessment to well-structured teaching
sessions and progressive introduction of higher values. Under these benign teaching conditions, we not
only capture the beneficial effects of past teaching but also create circumstances under which new
performances can emerge via spontaneous recombination – analogous somewhat to the benefits of
teaching phonics in reading instruction. These findings are directly in line with recent theory that suggests
that slow, inflexible learning of persons with intellectual disabilities may be avoidable through optimizing
aspects of instructional procedures.
Research activity supported by NICHD grants HD46319 and HD48144
68
14.
POSTER SESSION 1
Receptive Language in Adolescents with Fragile X Syndrome with or without Autism
S. T. Kover, L. Abbeduto, N. D. Giles, E. K. Richmond, P. Lewis, A. Weber,
S. Schroeder, J. A. Anderson, and A. Baron
Waisman Center, UW-Madison, 1500 Highland Avenue, Madison, WI 53705-2208
Contact: [email protected]
Introduction: There is an association between fragile X syndrome (FXS) and autism (AUT), with one-third
of individuals with FXS meeting criteria for AUT. It is unclear, however, whether individuals with FXS+AUT
are simply more severely affected than their FXS only peers or whether the two groups differ qualitatively.
Several studies have addressed this issue by comparing the linguistic profiles of FXS+AUT and FXS only,
but with inconsistent results. Roberts et al. (in press) found that boys (ages 3 to 14 years) with FXS and
an autism spectrum disorder did not differ in receptive language scores from boys with FXS only, Down
syndrome (DS), or typical development (TD), all matched on nonverbal mental age (NVMA). Roberts et al.,
however, measured only receptive vocabulary and did not distinguish children with AUT from others on the
AUT spectrum. In contrast, Lewis et al. (2006) found that adolescents with FXS+AUT (ages 11 to 23
years) scored lower than NVMA-matched adolescents with only FXS on measures of receptive vocabulary
and grammar. Lewis et al., however, relied on clinician judgment to make the diagnosis of AUT rather
than using the Autism Diagnostic Interview-Revised (ADI-R) and Autism Diagnostic Observation Schedule
(ADOS), which together are the gold standard for diagnosis. In the present study, we attempted to clarify
the receptive language phenotypes of FXS+AUT and FXS only by measuring both receptive vocabulary
and syntax, using the ADI-R and ADOS to diagnose AUT, and by including only participants with AUT in
the FXS+AUT group. We also included TD and DS comparison groups to provide a basis for determining
the extent of delay in receptive language in the two FXS groups.
Method: Participants were adolescents aged 10 to 15 years with FXS+AUT (n = 9, 8 males), FXS (n = 20,
18 males), DS (n = 20, 10 males), and TD children (n = 20, 11 males). Participants who met criteria on the
diagnostic algorithms for autism on both the ADI-R and the ADOS comprised the FXS+AUT group. All
groups were matched on NVMA, based on the Leiter-R. As part of a larger battery, participants received
the Peabody Picture Vocabulary Test (PPVT) and the Test for Reception of Grammar (TROG), a measure
of receptive syntax.
Results: Preliminary analysis of PPVT and TROG age-equivalent scores using separate one-way
ANOVAs revealed group differences on both measures. There were no differences between the
FXS+AUT and FXS only groups. Instead, participants with DS scored lower than participants with only
FXS on the PPVT, and all three syndrome groups scored lower than the TD group on the TROG. The
same findings emerged for raw scores and standard scores.
Discussion: Differences between adolescents with FXS+AUT or FXS were not found in receptive
vocabulary or syntax. Thus, there was no evidence of a qualitatively different profile in receptive language
(i.e., different profiles of strengths and weaknesses in receptive language relative to cognitive level)
between FXS+AUT and FXS only. These findings, which are contrary to the Lewis et al. study, could be
due to our differing language measures, improved diagnostics, or insufficient statistical power. Data from
additional participants and on group profiles across specific syntactic forms will be presented.
Lewis, P., Abbeduto, L., Murphy, M., Richmond, E., Giles, N., Bruno, L., et al. (2006). Cognitive, language
and social-cognitive skills of individuals with fragile X syndrome with and without autism. Journal of
Intellectual Disability Research, 50(7), 532-545.
Roberts, J., Price, J., Barnes, E., Nelson, L., Burchinal, M, Hennon, E. et al. (in press). Receptive
vocabulary, expressive vocabulary, and speech production of boys with fragile X syndrome in
comparison to boys with Down syndrome. American Journal on Mental Retardation.
69
15.
POSTER SESSION 1
Developmental Trajectories of Language Skills in Toddlers and
Adolescents with Down Syndrome
Nancy Raitano Lee1,2*, Susan Hepburn1, Amy Philofsky1, Deborah Fidler3,
Sally Rogers4, & Bruce Pennington2
1
JFK Partners; University of Colorado at Denver & Health Sciences Center; 4200 East 9th Avenue,
C268-30; Denver, CO 80262; 2Department of Psychology, University of Denver;
3
Department of Human Development & Family Studies, Colorado State University;
4
M.I.N.D. Institute, University of California–Davis
*Contact: [email protected]
Introduction: Research has suggested that the developmental trajectory of intelligence in individuals with
Down syndrome (DS) is characterized by slower rates of growth, such that gains in mental age (MA)
decrease as chronological age (CA) increases (Hodapp & Zigler, 1990). The current research sought to
augment our understanding of the developmental trajectory of language skills in DS by examining two
samples longitudinally, one in early childhood and one in adolescence.
Methods: The first sample included toddlers with DS (n = 8) and an MA-matched typically developing
control group (n = 8). Toddlers with DS were tested 2 years apart, while typically developing participants
were tested 1 year apart using the Mullen Scales of Early Learning. The second sample included
adolescents with DS (n = 15) who were tested approximately 7 years apart using the Peabody Picture
Vocabulary Test – Third Edition.
Results: For the MA-matched toddler sample, the relative gain in raw score points on the Expressive
Language scale of the Mullen was not similar across groups from Time 1 to Time 2. Even though the
typically developing control participants had only aged one year, their gain in raw score points on the
Mullen Expressive Language scale was significantly greater than the participants with DS who had aged
two years (Expressive = Main Effect Time: F(1,14)=85.35, p<.001; Trend for Group: F(1,14)=3.24, p<.1;
Group x Time Interaction: F(1,14)=4.66, p<.05; Receptive = Main Effect Time: F(1,14)=34.61, p<.001; No
effect of group; Trend for Group x Time Interaction: F(1,14)=2.75, p=.12). For the adolescent group,
because a control group was not followed over time, we relied upon norms from the PPVT manual using
two methods. First, we compared DS performance to 50th percentile raw scores matching on CA at Time 1
and Time 2. Second, we compared DS performance to 50th percentile raw scores matching on MA at Time
1 and then extrapolating age at time 2 (i.e., Time 2 age = Time 1 MA + time between the two testing
sessions). When using CA-matched norms, the proportional gain in raw score points in the DS group was
similar to that found for children with 50th percentile performance, despite expected group differences in
mean raw scores at the two age points (Main Effect Time: F (1, 28) = 58.51, p < .001; Main Effect Group:
F (1, 28) = 93.75, p <.001; No Group x Time Interaction: F < 2, p >.2). Similar to the toddler sample, when
using MA-matched norms at Time 1, a significant group x time interaction was found, such that the
adolescents with DS made a less significant gain in raw score points from Time 1 to Time 2 as compared
to the raw score norms associated with 50th percentile performance over that same period (Main Effect
Time: F (1, 26) = 165.76, p < .001; Main Effect Group: F (1, 26) = 8.41, p <.01; Group X Time Interaction:
F (1, 26) = 60.49, p <.001).
Discussion: Consistent with earlier findings, these results suggest slower rates of growth in language
development in DS when participants are compared to MA-matched controls. However, when adolescents
with DS in this sample were compared to CA-matched, 50th percentile norms, similar rates of growth in
receptive vocabulary skills over time were evidenced. Implications for choosing comparison groups for
longitudinal research will be discussed.
Key References: Hodapp, R. M. & Zigler, E. (1990). Applying the developmental perspective to individuals
with Down syndrome. In D. Cicchetti & M. Beeghly (Eds.), Children with Down syndrome (pp. 1-28). New
York: Cambridge University Press.
70
16.
POSTER SESSION 1
Mood and Anxiety in Mothers of Children with FXS and
Mothers of Children with Autism
Jean B. Mankowski1, B.A., Jane Roberts1, Ph.D. and Donald Bailey Jr2., Ph.D
FPG Child Development Institute at UNC-Chapel Hill1; RTI International2
Contact: [email protected]
Introduction: Parents of children with disabilities are faced with challenges such as the heightened
demand of childcare, continual questions concerning a child’s progression, physical and emotional fatigue
from daily care, and maladaptive or problematic child behavior. These challenges can impact a parent’s
mood, anxiety, stress level, and overall emotional well-being. Children with autism and children with fragile
X syndrome (FXS) are known to possess deficits in sociability, and they often exhibit maladaptive and
problematic behaviors. Autism and FXS are distinct disorders, but are related in a number of ways
including overlap in behavioral symptoms, neurological anomalies, functioning expressed within a range,
and differential patterns of genetic risk. Existing literature shows that female carriers of FXS and mothers
of children with autism display higher incidences of mood and anxiety disorders (Abbeduto, et al., 2004)
than found in community samples. However, prevalence rates have been inconsistent due to variations in
sample and instrumentation (screening vs. interview, current vs. past, diagnostic criteria). No study has
examined lifetime history of mood and anxiety using a structured interview based on the DSM-IV.
Additionally, no study has examined how maternal mood and anxiety have been affected by the birth and/
or diagnosis of the child with the disability, and/or the severity of the child’s autistic and difficult behaviors.
This study compares the history of mood and anxiety disorders in mothers of children with FXS, mothers
of children with autism (non-FXS), and mothers of children with FXS and autism.
Methods: Three participant groups are involved in this study: 1) 20 female carriers of FXS with a
biological son with elevated autistic behavior (CARSe”30), 2) 20 female carriers of FXS with a biological
son without elevated autistic behavior (CARS <30), and 3) 40 females with a biological son with autism
(non-FXS). The three groups are matched on adaptive behavior (Vineland Composite Score) and
chronological age. All carrier mothers of FXS have the premutation and their children have the full
mutation. Trained evaluators derive current and lifetime rates of anxiety and depression using The
Structured Clinical Interview (SCID) for the DSM-IV. Child measures include the Childhood Autism Rating
Scale, Vineland Adaptive Behavior Scale and Child Behavior Checklist. All data on the female carriers of
FXS and their children with FXS have been collected. Data from thirty-two mothers and their children with
autism have been collected, and up to ten more families will be assessed by November, 2006.
Results: Preliminary analyses of our data suggest a higher lifetime prevalence of major depressive
episodes (MDE) in mothers of children with FXS (42%, n=91) and mothers of children with autism (68%,
n=32) than in community samples (24%). Future analyses will examine how maternal mood and anxiety
are impacted by the severity of the child’s autistic and challenging behavior. In addition, we will examine
the attribution of major depression to critical life events including the birth and diagnosis of their child.
Discussion: These results will further our understanding of mood and anxiety in mothers of children with
FXS and mothers of children with autism and provide insight as to the extent that mood and anxiety
disorders may be psychiatric vulnerabilities in these populations. In addition, our findings will provide
information on the effect of child autistic and problem behavior on mood and anxiety disorders. These
findings can be used to guide discussion of the challenges families face and ways to provide more
informed support, intervention services, and treatment options for both mothers and their children.
Key Reference: Abbeduto, L., Seltzer, M.M., Shattuck, P., Krauss, M.W., Orsmond, G., & Murphy. M. M.
(2004). Psychological well-being and coping in mothers of youths with autism, Down syndrome, or fragile
X syndrome. American Journal on Mental Retardation, 109, 237-254.
71
17.
POSTER SESSION 1
Parents of Children with Autism Spectrum Disorders Have Merited Concerns
About Their Infant Siblings
Caitlin R. McMahon, Elizabeth E. Malesa, Paul J. Yoder, & Wendy L. Stone
Vanderbilt University, Psychology and Human Development, & Vanderbilt Kennedy Center
Email: [email protected]; Mail: 1500 21st Ave S, Suite 4200, Nashville, TN 37212
Introduction: Infant siblings of children with ASD (SIBs-ASD) are at elevated risk for social and language
delays (Landa & Garrett-Mayer, 2006; Yirmiya et al., 2006). Little is known about how parents perceive
the development of Sibs-ASD. It is possible that parents may be hypervigilant (i.e., excessively worried)
about noting concerns in the development of their younger siblings. Thus, the extent to which parental
concern is related to true tested cognitive or language impairment in the SIBs-ASD is unclear. The aims
of the current study were to test: (a) the between-group differences (SIBs-ASD vs SIBs-TD) on parental
concerns of their infant’s development, and (b) the association between the extent to which parents
express developmental concern and their infants’ standard scores on an early developmental scale.
Method: Seventy-six participated in the study: 49 Sibs-ASD (mean Chronological age (CA)= 15.9; range=
12-24 months) and 27 CA-matched siblings of typically-developing children (SIBs-TD; mean CA= 16.1,
range= 12-23 months). Parents completed the Parental Concerns Form (PCF) and each child received
the Mullen Scales of Early Learning (MSEL). The PCF includes 7 questions rated on a 3 point scale (i.e.,
“no=0,” “a little=1,” “yes=2”), that address concerns about the child’s language use, language
understanding, use of hands and fingers, use of arms and legs, behavior, ability to get along with others,
and learning. The Total Score from the PCF (range 0-14) was used for analyses and tested for
associations with MSEL Language Domain T-scores and overall MSEL Early Learning Composite (ELC)
standard score.
Results: Groups did not differ on CA or gender. The Sibs-TD group had a significantly higher mean level
of maternal education than the Sibs-ASD group (t(74) = -2.62, p = .011; Cohen’s d=.60). Maternal
education was negatively correlated with Total Parental Concerns (r = -.287, p = .01) and was positively
correlated with MSEL Receptive Language T-score (r = .315, p = .006). Therefore, maternal education
was entered as a covariate in analyses as appropriate. ANCOVA analyses revealed that groups differed
significantly on overall parental concerns (F(1, 73)= 11.12, p= .001, partial eta squared= .132), with
parents of Sibs-ASD indicating more total concerns than parents of Sibs-TD. Sibs-ASD received
significantly lower scores than Sibs-TD on overall MSEL ELC score (F(1,74)= 10.36, p=.002, Partial eta
squared=.123). No differences emerged between groups on MSEL Receptive Language after controlling
for maternal education (F(1,73)= 2.31, p=.133, Partial eta squared=.031) and no differences between the
groups were found for MSEL Expressive Language (F(1,74)= 2.57, p=.113, Partial eta squared=.034).
Regression analyses revealed that each of the MSEL scores of interest independently predicted total
parental concerns; MSEL ELC standard score (t= -4.91, p= .000), MSEL Receptive Language T-score (t= 2.43, p= .018), and MSEL Expressive Language T-score (t= -4.47, p= .000). Parental concerns remained
significantly and negatively correlated with MSEL Total ELC standard score (r = -.383, p=.001) and with
MSEL Receptive Language T-score (r = -.280, p=.015) when MSEL scores were split dichotomously at
the 10%ile, suggesting that parental concerns are justified by their relation to clinically significant delays.
Discussion: Parents of children with ASD are more concerned about the development of their youngerborn siblings than are parents of typically developing children. Our results suggest that these concerns are
warranted in that higher parental concerns were related to lower standard scores on cognitive and
language measures. The validation of parent concerns has implications for parents and professionals in
seeking specialized intervention services for younger siblings.
Key References:
Landa, R. & Garrett-Mayer, E. (2006). Development in infants with autism spectrum disorders: a
prospective study. J Child Psychol Psychiatry, 47(6), 629-638.
Yirmiya N, et al. (2006). The development of siblings of children with autism at 4 and 14 months: social
engagement, communication, and cognition. J Child Psychol Psychiatry, 47(5), 511-23.
72
POSTER SESSION 1
18.
Personality, Coping Style, and Adjustment of Parents Rearing Children with
Developmental Disabilities
Angela L. Natcher, Brian M. Jobe, and Laraine M. Glidden
Department of Psychology, St. Mary’s College of Maryland, St. Mary’s City, MD 20686
Contact: [email protected]
Parents rearing children with developmental disabilities (DD) encounter stressors that require coping and
adaptation. In previous research, the use of problem-focused coping strategies was more often
associated with positive adjustment outcomes than was the use of emotion-focused coping. Glidden,
Billings, and Jobe (in press) reported this pattern with regard to depression and subjective well-being
measured concurrently with coping. However, Positive Reappraisal, an emotion-focused coping
mechanism, was an exception, predicting greater well-being. In addition, parental personality was shown
to influence outcomes, with Neuroticism, in particular, associated with lower well-being. The current study
aims to replicate these results for the same adjustment outcome variables measured 5.5 years later.
Additionally, we hypothesized that, in regard to the child’s transition to adulthood, use of problem-focused
coping strategies would predict less worry and emotion-focused coping strategies (with the exception of
Positive Reappraisal) would predict more worry.
For the present study, we examined the DEP5, a 5-item measure of depression, Subjective Well-being
(SWB) relating to Global, Current, and Child-related functioning, as well as the Transition Daily Rewards
and Worries Questionnaire (TDRWQ), a self-report inventory used to assess parental reward and worry
concerning the child’s transition into adult life. Participants included 138 mothers and 69 fathers parenting
at least one child with DD. In hierarchical regression procedures for each criterion variable, parental role
(mother/father) and the five personality factors from a self-report NEO were entered as a block before the
eight coping strategies from the Ways of Coping (WOC) questionnaire were entered in a stepwise
procedure. NEO and WOC data had been collected approximately 5.5 years earlier.
In none of the analyses did mother/father differences reach significance, indicating no significant main
effect of parental role for any of the criterion variables. Higher scores on Neuroticism predicted greater
depression and lower global and current SWB, replicating the earlier findings of Glidden, Billings, and
Jobe. The findings suggest that, even after controlling for personality, the coping strategies used by the
parents of children with DD are predictive of their well-being outcomes more than 5 years later. In general,
problem-focused coping strategies were more effective than were emotion-focused coping strategies (with
the exception of Positive Reappraisal) at achieving positive adjustment outcomes. Specifically, Planful
Problem Solving and Positive Reappraisal predicted less parental worry concerning the child’s financial
independence and Self-controlling and Accepting Responsibility coping strategies predicted more parental
worry. The use of Confrontive Coping, a problem-focused coping strategy, was associated with lower
depression and greater Global and Child-related SWB, whereas Escape-avoidance, an emotion-focused
coping strategy, was associated with greater depression and lower Child-related SWB. Similarly, the
emotion-focused strategies, Self-controlling and Distancing, predicted greater depression and lower
Global SWB, respectively. Additionally, all emotion-focused coping strategies except Positive Reappraisal
predicted increased parental worry concerning various aspects of the child’s transition into adulthood.
These findings corroborate earlier ones and demonstrate consistency with intervention programs that aim
to alter individual coping style for more effective adjustment to stress.
Reference
Glidden, L. M, Billings, F. J., & Jobe, B. M. (in press). Personality, coping style, and well-being of parents
rearing children with developmental disabilities. Journal of Intellectual Disability Research.
73
19.
POSTER SESSION 1
The Effect of Disability Status and Social Experience on Social Cognitive Development
Irene Ngai & Frank J. Floyd, Georgia State University
Janis B. Kupersmidt, University of North Carolina at Chapel Hill
Contact: Irene Ngai 1376-3 Benning Place NE Atlanta, GA 30307
[email protected]
Social cognitive skills enable children to make sense of and respond to incoming social information.
Social cognitive development is influenced by both general cognitive functioning and social experiences.
Thus, because of cognitive delays, children with Down syndrome (DS) and children with mental retardation
of unspecified etiology (MR) are expected to demonstrate delays in their social cognitive skills compared
to children without disabilities. However, social cognitive development might vary between the disability
groups according to differences in social experience. The present study examined the effect of the child’s
diagnosis and social experiences on the development of two social cognitive skills, interpretation of peer
intent and response strategy generation. Specifically, children with DS were expected to experience more
prosocial treatment by peers and less physical and relational aggression than the children with MR. Thus,
children with DS were expected to make fewer hostile attributions, and consequently, suggest more
prosocial and fewer retaliatory response strategies than the children with MR. Yet, as the sample aged,
increasing awareness of social norms was expected to contribute to a decline in hostile attributions and
retaliatory responses, and an increase in prosocial responses.
The present study included data for 18 children with MR and 9 children with DS. To assess age-related
changes in social cognitive skills, up to 4 waves of data were obtained using the Social Problem Solving
Interview (Dodge, Pettit, McClaskey, & Brown, 1986). This interview included three hypothetical social
scenarios – one involving exclusion (e.g., child’s request to join a play group is rebuffed), one involving an
ambiguous aggressive incident (e.g., drink is spilled on the child), and one involving social aggression
(e.g., children whispering near the child). We examined the percentage of hostile interpretations proposed
as well as both the proportion of prosocial and retaliatory response strategies suggested. The Social
Experiences Questionnaire (Crick & Grotpeter, 1995), was used to obtain parent reports of the child’s
experiences as a recipient of prosocial behaviors and as a victim of both overt and relational aggression.
Sixteen items reflected prosocial experiences (e.g., “my child has peers who like to play with him/her”),
overt aggression (e.g., “my child is in danger of being hit by others”), and relational aggression (e.g.,
“other children prevent my child from joining an activity”).
Hierarchical linear modeling was used to investigate the impact of disability status and social experience
on the development of two social cognitive skills. Contrary to expectations children with DS suggested
more hostile interpretations of peer intent than children with MR. However, consistent with expectations as
to the effects of social experience, children with DS who were the recipient of low levels of prosocial
behavior, and who experienced the most physical and relational aggression demonstrated the greatest
increases in hostile interpretations over time. Regarding response strategies, regardless of their social
experience, the mean number of retaliatory strategies suggested by children with DS was significantly
higher than that of other children with MR. Moreover, with increasing age, children with DS who experienced a low level of relational aggression showed the slowest growth in their use of prosocial responses.
Overall, the findings suggest that for children with DS, negative social experiences contribute to maladaptive cognitions about peer intent, which in turn, influence the types of social responses suggested. Moreover, the absence of maltreatment was not associated with prosocial tendencies. This pattern of responses continued with increasing age. These findings are interpreted as evidence that children with DS
develop attribution and response tendencies based on past social experiences, and that cognitive delay
may inhibit growth for these skills. Future researchers are encouraged to recruit larger samples of children with MR and DS to further explore inter-group differences found in the present study. Researchers
may also study how social cognition and social experience interact to impact social adjustment over time.
74
20.
POSTER SESSION 1
Emotional and Behavioural Adjustment in Siblings of Children with Intellectual Disability
with and without Autism
Michael A. Petalas, Susie Nash, and Tracey Lloyd
School of Psychology, University of Wales, Bangor
Contact: [email protected]
Introduction: Siblings of children with autism have often, but not always, been found to present with more
adjustment difficulties in comparison to siblings of children with other disabilities or siblings of typically
developing children. The main aims of the present study were: 1. To compare the behavioural adjustment
of siblings of children with autism and intellectual disability (ID) with adjustment in siblings of children with
ID, and a normative sample; and 2. To examine factors that may be related to the adjustment of siblings of
children with autism. In addition to a cross-sectional exploration of these questions, data were gathered for
an 18 month follow-up of these siblings.
Method: Mothers rated the adjustment of the sibling closest in age to the child with ID with or without
autism. Data were available on 13 brothers and 13 sisters of siblings of children with autism (mean age
[SD] = 10.4 [3.3]), and 18 brothers and 9 sisters of children with ID (mean age [SD]= 11.3 [3.5]). Mothers
completed the Strengths and Difficulties Questionnaire (SDQ), a standardized measure of behavioural and
emotional adjustment for children and adolescents, and a number of other measures about themselves
and their family. Data are available for 17 siblings of children with autism followed up 18 months later.
Results: Siblings of children with autism were rated as having more emotional problems when compared
with siblings of children with ID and also with a normative sample of British children. No other dimensions
of adjustment showed group differences. Siblings of children with autism who were younger than the child
with autism, had a sister with autism, and whose sibling was older had more reported emotional problems.
Socioeconomic deprivation was associated with higher levels of reported emotional and behavioural
adjustment difficulties. Over 18 months, behavioural and emotional adjustment (with the exception of
problems with peers) was stable for the siblings of children with autism (r values >.70).
Conclusions: These data suggest that adjustment problems in siblings of children with autism and ID may
be confined to emotional difficulties. Although a number of correlates of emotional problems were also
found, the mechanism for these increased emotional problems is not clear and should be explored in
future research.
75
21.
POSTER SESSION 1
Health Knowledge and Expected Outcomes of Risky Behaviour: A Comparative Study of
Non-Disabled Adolescents and Young People with Intellectual and Physical Disabilities
Pownall, J*, Jahoda, A, Wilson, University of Glasgow, Scotland, UK.
*Section of Psychological Medicine, Division of Community Based Sciences, Gartnavel Royal Hospital,
University of Glasgow, Glasgow, G12 0XH, UK
Contact: [email protected]
Introduction: The physical health needs of people with intellectual disabilities are increasingly being
advocated. Unfortunately there is an absence of research focusing on young people with intellectual
disabilities in relation to their understandings about health and the factors that promote or maintain healthrisking behaviours. However, there is often an assumption that a lack of health understanding arises from
their cognitive deficits. The present study adopted a group comparison design in order to examine the role
played by social exclusion in the acquisition of health knowledge. As young people with intellectual and
physical disabilities are more likely to experience social exclusion, the opportunities they have to talk about
health may be fewer and this in turn may impact upon their knowledge. It was hypothesised that gaps in
knowledge would be more marked for private and sensitive topics like sexual health, which are discussed
far less openly than other more public topics such as healthy eating or alcohol.
Methods: Seventy-six young people aged between 15-21 participated in the study. The health knowledge
of the following three groups of young people were compared: (i) 34 with no known disability (ND), (ii) 34
with a mild/moderate intellectual disability (ID), and (iii) 28 with a physical disability (PD). Questionnaires
and semi-structured interviews were used to elicit both quantitative and qualitative data regarding
knowledge of two public health areas (healthy eating and alcohol consumption) and two private areas
(contraception and AIDS). Information was also collected regarding participants’ social networks and
sources of health knowledge.
Results: Chi-square tests revealed that young people with ID had poorer health knowledge overall when
compared to the ND and PD groups. However, with regard to contraception, both the ID and PD groups
performed similarly. As the PD group had no intellectual impairment, this suggests that such knowledge
deficits were a result of social exclusion as opposed to an ability to understand and retain information per
se. Young people with disabilities also had significantly fewer social contacts in and out of school/college
and reported having fewer opportunities to discuss and access information regarding sexual health than
their non-disabled peers.
The qualitative data also revealed major misunderstandings in relation to sexual health across groups,
although this was more pronounced for the ID and PD groups.
Discussion: Young people have a good grasp of key health messages in relation to healthy eating and
alcohol consumption. For more private health topics like safe sex it appears that limited opportunities to
socialise with peers and engage in age normative tasks, and poorer access to other sources of
information is negatively impacting upon knowledge. If we are to empower young people to make sensible
and informed decisions about their health, we must take into account their lay theories of health, including
both understandings and misunderstandings. By starting with people’s own meanings of health and illness
we are better positioned to design effective education strategies.
.
76
22.
POSTER SESSION 1
Parental Coping Assistance with Peer Problem Solving for Children
with Intellectual Disabilities
Kara Snead1, Frank J. Floyd1, & Janis Kupersmidt2
Georgia State University, 2University of North Carolina at Chapel Hill
Contact: Kara Snead Georgia State University PO Box 5010 Atlanta, GA 30302
[email protected]
1
Children with intellectual disabilities experience problems with peers that are especially difficult for them to
manage. Parents are likely a primary source for learning skills to cope with peer problems. At present,
there is lack of research concerning parental coping assistance for children with intellectual disabilities.
Compared to parents of children without disabilities, parental coping assistance might differ because of
varying perceptions of the children’s abilities and needs and the child’s role in the problem. This study
explored how parents of children with intellectual disabilities assisted their children with managing peerrelated problems. We expected that, compared to other parents, those who have children with intellectual
disabilities would perceive their children as having less control over the peer problem and, thus, would
tend to suggest or provide passive, palliative coping assistance rather than active strategies, or would
directly manage the problem for the child.
Participants included 136 families (including both mothers and fathers, when present) with children, 8-10
years old, in three groups: children with mild or moderate mental retardation or Down syndrome (MR
group, n = 44), children with learning disabilities (LD group, n = 60), and children who are typically
developing (Comparison group, n = 32). Parents were interviewed separately using the Parental
Socialization of Coping interview (Kupersmidt, Clarke, & Morey, 1997). During the interview, the parent
described a recent peer problem experienced by the child and answered a series of questions about
perceived control by the child over the stressor occurring in the first place and continuing once it began,
and control over the outcome. The parents also described their efforts to help the child cope with the
problem, and reported on other direct activities by the parent toward solving the problem. Finally, they
indicated whether the coping efforts were effective and whether the problem was resolved successfully.
For this analysis, coping suggestions and activities were classified as active coping assistance, which
included offering advice, suggesting behavioral strategies, aiding the child in analyzing the problem, and
engaging in active problem-solving with the child. Passive coping assistance included suggesting cognitive
avoidance, listening and offering emotional support, and suggesting tension reduction strategies.
Initial data analyses indicated that as expected, mothers of children with MR tended to view their children
as having relatively little control in the peer situation. However, these mothers provided the least amounts
of both passive and active coping assistance, overall. When coping assistance was offered, mothers of
children with MR tended to provide fewer passive suggestions and more active suggestions in situations of
low control. In comparison, father’s of children with MR made active suggestions during situations of high
control and passive suggestions during situations of low control as predicted. Across groups, fathers’
perceptions of child control and coping suggestions did not differ. Contrary to expectations, mothers and
fathers engaged in direct parental involvement regardless of their child’s disability status.
These findings suggest that the type of coping assistance offered by fathers of children with MR was
consistent with perceptions of their child’s control. In contrast, mothers of children with MR provided
coping assistance that was incompatible with their perceptions of their child’s control. Although a positive
consequence may be that these mothers provide their children with a sense of agency, this may also lead
to frustration and confusion for children in uncontrollable peer situations. Therefore, future research should
examine the implications of these parent behaviors on social functioning and subsequent peer-related
skills for children with MR.
77
23.
POSTER SESSION 1
Is Maternal Responsivity Different in Mothers of Young Children with Fragile X Syndrome
Compared to Down Syndrome?
Audra Sterling, Nancy Brady, and Steven F. Warren
University of Kansas
1000 Sunnyside Blvd., Room 1052,
Lawrence, KS 66045;
[email protected]
Parenting style can have a profound impact on the development of young children. For example, maternal
responsivity,has been found to impact children’s cognitive, emotional, and language development (Landry,
Smith, Miller-Loncar, & Swank, 1998; Landry, Smith, Swank, Assel, & Vellet, 2001). A mother who is highly
responsive typically maintains her child’s focus of attention, expands on the child’s initiations, and rarely
redirects the child to a new topic. Both fragile X syndrome (FXS) and Down syndrome (DS) present a
number of unique challenges that may affect parenting style, including delays in language and
communication, and behavioral and attentional problems. Differences associated with the two disorders
may differentially affect responsivity, however. Women who are carriers of FXS or who have the full
mutation are susceptible to depression and social anxiety, which could affect maternal responsivity.
Mothers of children with DS are typically aware of their child’s disability much earlier than mothers of
children with FXS and may have developed adaptive interaction styles based on early identification. For
this poster presentation, we compare maternal responsivity of mothers of children with DS to mothers of
children with FXS.
Method: We compared 12 mothers of children with DS to 12 mothers of boys with FXS. The children in
both groups were matched on both expressive language raw scores from the Mullen Scales of Early
Learning (MSEL; mean score = 16.7 for DS, 16.25 for FXS), and on chronological age (mean = 33.75 mo.
for DS; 32.3 mo. for FXS). Mother-child interactions were coded from videotapes of three contexts: free
play, book reading, and making a snack.
Results: A factor analysis was completed in order to obtain two factors for maternal responsivity. Behavior
Management consisted of redirects, zaps and request for behavioral complies. Facilitative Interaction Style
was made up of gestures, recodes, request for verbal complies, and comments. We ran t-tests in order to
evaluate the differences between the two groups on each factor. The two groups were significantly different on the Behavior Management factor (t=2.634, p=.015), but were not significantly different on the
Facilitative Interaction factor (t=.100, p=.921). Follow up analyses indicated that mothers who have a child
with DS redirected their children significantly more often than mothers of children with FXS (t=10.834,
p=.00), and this difference seems to account for the difference in the Behavior Management factor.
Discussion: The finding of significant group differences in the Behavior Management factor was surprising because the children were well-matched on language and chronological age. Research by Legerste,
Varghese & Beek (2002) and Marfo (1990) found that language level accounted for differences in maternal
interaction style. However, our findings are consistent with those of Mahoney (1988) who found mothers of
children with DS were more directive than mothers of children without disabilities. Results should be
interpreted with caution, however, due to small sample sizes.
Acknowledgement: This research is supported in part by grants 3 P30 HD003110-3 and P30 HD00252839, from NICHD.
78
24.
POSTER SESSION 1
The Effects of Prematurity on Concept Formation at Five Years of Age
Vicki Sudhalter, Richard C. Belser, Judith M. Gardner and Bernard Z. Karmel
N.Y.S. Institute for Basic Research in Developmental Disabilities
1050 Forest Hill Road, Staten Island, New York 10314
[email protected]
Introduction: Continuing advances in prenatal and postnatal care have led to an increased survival rate
of infants born prematurely, or with various other perinatal risk factors. It has been suggested that the
effects of prematurity may continue beyond the neonatal period, and interfere with later neurocognitive
development. (Aylward, 2005; Salt & Redshaw, 2006). The data being presented here address the
relationship between prematurity and the development of concept formation skills during the early school
years.
Methods: Our preliminary sample includes 32 children with a mean gestational age of 36 weeks, ranging
from 29 to 41 weeks. They had a mean birth weight of 2732 grams, ranging from 1389 to 4479 grams.
Each child was administered the Children’s Category Test (CCT, The Psychological Coportation,1993),
according to the standard testing protocol. The CCT is designed to assess non-verbal learning, memory,
concept formation and problem solving abilities. We used the Level I version of the CCT, which is
designed for children between the ages of five and eight years. Each child was shown a stimulus booklet
containing colored images of geometric shapes, and a color response card, and was asked to “point to the
color that you think of when you look at the picture.” There are four subtests of increasing difficulty, and a
fifth subtest which reviews the concepts of the first four.
Results: The data from our preliminary sample were examined by multiple regression analysis (using
Statistica) to determine the influence of prematurity and age upon CCT performance. The contribution of
prematurity (i.e., estimated gestational age at birth) to the CCT standard score was significant [beta =
0.41, p=.02], and its contribution to the Subtest IV score (which includes the most difficult concepts) was
also significant [beta = 0.42, p=.02]. The contribution of age at testing was not significant in either case.
Discussion: Our preliminary data suggest that there is an important relationship between prematurity and
later problems in concept formation abilities. Successful performance on the CCT also requires the ability
to change problem solving strategies, to develop alternative solutions, and to benefit from experience.
Thus, it directly assesses the cognitive processes required for successful academic achievement. By
enhancing our understanding of the relationship between prematurity and such cognitive processes, we
will be better able to identify those children at greatest risk for academic problems, and to target remedial
resources to those at-risk children who would most benefit from them.
Key References:
Aylward, G. P. (2005). Neurodevelopmental outcomes of infants born prematurely. Developmental and
Behavioral Pediatrics. 26, 427-440.
Salt, A. & Redshaw, M. (2006). Neurodevelopmental follow-up after preterm birth: follow up after two
years. Early Human Development, 82, 185-197.
79
25.
POSTER SESSION 1
Interactive Training on Active Support: Perspectives from Staff, Managers, and Trainers
Vaso Totsika, Sandy Toogood, Susie Nash
University of Wales, Bangor
Vaso Totsika, School of Psychology, University of Wales, Bangor, Adeilad Brigantia, Penrallt Road,
Bangor, Gwynedd, LL57 2AS
[email protected]
Introduction: Active Support (AS) is an organizational technology for people with intellectual disabilities
living in small community homes. AS aims to maximize opportunities for people to participate fully in
everyday, life-defining activity and engage in meaningful social interaction. Staff working in community
homes receive training in AS through group workshops and in-house Interactive Training (IT). Although the
effectiveness of AS for people with an intellectual disability has been established, there are no existing
data on the experience of staff trained to use AS. This study reports on the IT experience of staff,
managers and trainers.
Methods: Fifty-eight staff received IT on AS in 10 residential houses. Nine months after the training, semistructured interviews were conducted with 37 staff, 6 house managers, 5 service managers and 6 trainers.
The interviews focused on people’s experience of the IT, the way it affected their everyday work and the
way the AS model is implemented in the houses they work.
Results: Preliminary analyses indicate that for about 90% of staff IT was a positive experience. The
opportunity for one-to-one training was the most frequently reported (about 23%) helpful characteristic of
IT by staff and managers. Other helpful IT factors included being trained by someone who is not a
member of the same staff group (about 17%) and the immediacy with which feedback is given and skills
are practiced and corrected (about 17%). The most frequently reported difficulties, when doing IT in a
service, were being observed by the trainers (about 30%), organizing the training session (26%) and the
difficulties that residents have in participating in an activity (26%). The majority of staff and managers
(about 65%) identified at least one new skill people use in their everyday work after IT, and about 25%
reported that IT has increased their awareness about support roles and resident needs. Preliminary
analysis of the staff and managers’ reports with regard to the way the AS model works in the houses
indicates that the implementation was incomplete, with some structural elements of AS completely missing
(e.g. teaching plans) and some only partially used (e.g. opportunity plans). Although about 30% of the
sample reported that AS was put in place to improve consistency of support, staff could not easily identify
the AS structural elements they use and there are indications of discrepancies in the terminology used to
refer to them both within and across houses.
Discussion: Staff views on IT and AS training is an under-researched area. Findings from the present
study will be discussed in relation to the social validity of the intervention and to factors related to the
implementation and maintenance of the model in a community residential service. The findings have
implications for the design and implementation of staff training on AS in real-world settings.
Key Reference: Jones, E., Perry, J., Lowe, K., Allen, D., Toogood, S., & Felce, D. (1996). Active Support:
A Handbook for Planning Daily Activities and Support Arrangements for People with Learning Disabilities.
Booklets 1-6. Cardiff: Welsh Centre for Learning Disabilities Applied Research Unit.
80
26.
POSTER SESSION 1
Life Events and Psychological Outcomes in Adults with Intellectual Disabilities:
Longitudinal Relationships
Lee Williams*, Richard Hastings, Stephen Noone, Lauren Burns, Jessica Day, John Mulligan
School of Psychology, University of Wales Bangor,
Brigantia Building, Penrallt Road,
Bangor, Gwynedd, LL57 2AS, United Kingdom
Introduction:
In recent years there has been increasing interest in significant life events as a factor in the aetiology of
psychological disorders and challenging behaviour in people with intellectual disabilities. To date,
measures have comprised a list of events categorized as negative a priori by clinicians or researchers.
Studies using these measures have produced associations of modest effect size. Esbensen & Benson
(2006) have suggested that better predictive power may be achieved if the positive or negative impact of
life events is considered in the context of the individual’s life.
Methods:
The Bangor Life Events Schedule for Intellectual Disabilities Informant version (BLESID-I) comprises a list
of 38 events, each rated on two dimensions: frequency (never, once, or more than once) and impact (5point scale from considerable negative impact to considerable positive impact). The BLESID-I was
completed with carers of 89 people across the range of intellectual disability (preliminary data here; N=40).
The current study was longitudinal with a three-year time lag to address issues relating to the direction of
causality. Outcome measures of anger, aggression, physical and mental health and adaptive behaviours
were taken.
Results:
A scoring technique was developed based on the approach of defining life events as negative in the
context of the participant’s life. This system of scoring produced stronger associations with the
psychological measures and was thus taken forward into further analyses. Five regression models were
tested, each predicting a measure of outcome from negative life events, controlling for an initial levels on
the outcome measure. The negative life event measure was found to significantly predict anger (β = .34,
p = .03), aggression (β = .50, p < .01), affective/neurotic symptoms (β = .50, p < .01), and psychotic
symptoms (β = .54, p < .01). The prediction of adaptive behaviour approached significance (β = .19, p =
.05). No significant associations were found between earlier measures of adjustment and subsequent life
events, suggesting a uni-directional relationship in this sample.
Discussion:
We have provided further evidence for the importance of significant life events in the aetiology of
psychological disorder and challenging behaviour. The associations are generally stronger than those
found elsewhere in the literature, suggesting that the BLESID represents an improvement in life event
measurement in the ID population. Further data are needed to address the reliability of the BLESID,
however, completion of the BLESID with a carer may provide a quick way to provide evidence for or
against a number of differential diagnoses in a given presentation.
Esbensen, A. J., & Benson, B. A. (2006). A prospective analysis of life events, problem behaviours and
depression in adults with intellectual disability. Journal of Intellectual Disability Research, 50(4),
248-258.
81
Symposium 7
S Y M P O S I U M
7
Selective Neuropsychological Changes Across the Adult
Lifespan in Down Syndrome
Chair:
Darlynne A. Devenny
New York State Institute for Basic Research in
Developmental Disabilities
Discussant: A.J. Holland
University of Cambridge
82
SYMPOSIUM 7
Selective Neuropsychological Changes Across the Adult Lifespan in Down Syndrome
Chair:
Darlynne A. Devenny
New York State Institute for Basic Research in Developmental Disabilities
Discussant: A.J. Holland
University of Cambridge
Declines in Visuo-spatial Abilities in Young Adults with Down Syndrome
D.A. Devenny, P. Kittler, S.J. Krinsky-McHale
New York State Institute for Basic Research in Developmental Disabilities
Visuo-Spatial Working Memory Decline Associated with MCI and DAT
S.J. Krinsky-McHale1 ,P. Kittler1,W. Silverman2,D.A. Devenny1
1
New York State Institute for Basic Research in Developmental Disabilities
2
Kennedy Krieger Institute and John Hopkins University School of Medicine
Neuropathological Evidence of Alzheimer’s Disease in Down Syndrome
L.D. Nelson, V. Kepe, P. Siddarth, J. Barrio, G. Small
University of California Los Angeles
83
SYMPOSIUM 7
Declines in Visuo-Spatial Abilities in Young Adults with Down Syndrome
D.A. Devenny, P. Kittler, S.J.Krinsky-McHale
New York State Institute for Basic Research, Staten Is. New York
Contact: [email protected]
Introduction: Down syndrome (DS) is the most common genetic condition leading to a developmental
disability, with recent studies showing that aging occurs prematurely. Very little is known about cognitive
development across the adult lifespan of individuals with DS because studies of age-associated decline
rarely include young adults (Devenny et al., 1996; Hawkins et al., 2003; Oliver et al., 1998). Down syndrome
is associated with a specific cognitive profile that includes a relative strength in visuo-spatial processing and
a relative weakness in auditory working memory and central executive functioning. However, there are no
studies with a broad enough age range to examine how the unique set of abilities associated with the behavioral
phenotype of DS interacts with normal aging processes.
Method: All participants had DS, with full trisomy 21 confirmed by cytogenetic testing. Participants older
than 35 years have been followed with neuropsychological measures in a longitudinal study and none have
shown the declines or symptoms associated with Alzheimer’s disease. IQ scores across groups were
comparable (Table 1.) We compared performance on three working memory visuo-spatial tasks (1. Corsi
Block-tapping Test forward and backward; 2. Table Top Test involving spatial but not sequential memory; 3.
Visual Sequential Memory Test (adapted from the Illinois Test of Psycholinguistic Abilities) with three working
memory auditory tasks (1. Digit span forward and backward; 2. Word span; 3. Sentence Repetition).
Performance on each task was analyzed with an ANCOVA with age-group as a between-subjects factor and
IQ as a co-variate.
Results: Significant differences between age-groups were found
Table 1
for each of the visuo-spatial working memory tasks, except for
Age-Group N
Age
IQ
the Corsi span forward. Post hoc paired-comparisons showed that
participants younger than 25 years performed significantly better
15 – 24 yrs
8
19.7 + 3.1 59.2 + 7.2 than all other groups on Corsi span backward. On the Table Top
Test, participants younger than 35 years were better than
10
29.9 + 4.3 56.9 + 6.6 participants in the two older groups. On the Visual Sequential
Memory Test participants younger than 35 years were better than
35 – 44 yrs
22
40.0 + 2.8. 59.8 + 8.0
individuals older than 44 years, but were not significantly different
from those in the 35-44 yr decade In contrast, there were no
45 – 54 yrs
24
49.4 + 3.2 52.7 + 9.0 significant age-group differences on the auditory working memory
tasks, although Sentence Repetition approached significance (p
14
= .052) with small declines observable across the age-groups.
12
Cor s i Span Back w ar d
10
8
6
4
2
0
15 - 2 4
25- 34 35- 44 45- 54
A ge Gr oup
Discussion: This study confirms a previous, preliminary finding
that visuo-spatial working memory declines early in adult life in
adults with DS (Devenny et al., 1999). Since visuo-spatial
processing is an area of cognitive strength, these declines are
likely to have an important impact on overall cognitive organization
in these individuals. Similar declines in auditory working memory
were not observed, indicating that the differences across age
groups cannot simply be interpreted as a cohort effect. While the
basis of these selective declines are unknown, it may be that the
early accumulation of amyloid associated with the third copy of
chromosome 21 subtly influences neural processing in the
prefrontal cortex and its connection with the visual pathways.
Devenny, D.A., Krinsky-McHale, S.J., & Kittler,P. (1999, March). Paper at 32nd Annual Gatlinburg Conference,
Charleston, SC.
Devenny, D.A., Silverman, W.P., Hill, A.L., Jenkins, E., Sersen, E.A., & Wisniewski, K.E. (1996). JIDR, 40, 208-221.
Hawkins, B.A., Eklund, S.J., James, D.R. & Foose, A.K. (2003). Mental Retardation, 41, 7-28.
Oliver, C., Crayton, L., Holland, A., Hall, S. & Bradbury, J. (1998). Psychological Medicine, 28, 1365-1377.
This work was supported by funds from the NYS Office of Mental Retardation and Developmental Disabilities and by
grants from the NIH AG 14771 (Devenny) and HD 35897 (Silverman).
84
SYMPOSIUM 7
VisuoSpatial Working Memory Decline Associated with MCI and DAT
1
S.J. Krinsky-McHale1, P. Kittler1, W.Silverman2, & D.A. Devenny1
New York State Institute for Basic Research in Developmental Disabilities, Staten Island, NY
2
Kennedy Krieger Institute and John Hopkins University School of Medicine
Contact: [email protected]
Introduction: Visuo-spatial working memory has been found to be impaired in individuals with dementia
of the Alzheimer Type (DAT) in the general population (2). It has also been found to be a relative strength
in healthy, young individuals with Down syndrome (DS) (4). It is therefore crucial to understand the fate of
this aspect of working memory with the cognitive decline associated with normal aging, mild cognitive
impairment (MCI), and DAT.
Participants: The current analyses involved a subset of 59 participants with DS from our larger study;
13 participants were in early-stage DAT and had received a diagnosis from a physician (M age=56.92
±5.12; M IQ=55.31 ±5.12); 14 participants were classified as exhibiting MCI because they have shown
significant memory decline without observed changes in adaptive functioning (M age= 54.21±5.27 yrs, M
IQ=48.14 ±10.24); and 32 participants have remained healthy (M age =53.79 ±5.94, M IQ=53.03 ±13.02).
All participants had full trisomy 21 confirmed by cytogenetic testing.
Procedure: Two measures of visuo-spatial working memory were
administered, (a) the forward span of the Corsi Block-Tapping Test (5)
reflecting the spatial component of visuo-spatial working memory by assessing
the ability to reproduce a series of block taps in the correct sequential order,
and (b) The Visual Sequential Memory Test (VSM), an adaptation of the Illinois
Test of Psycholinguistic Abilities (3) reflecting the visual component of visuospatial memory by assessing the ability to reproduce a pattern of 2-dimensional
non-meaningful figures from memory. For both measures the score was the
length of the last sequence that was correctly reproduced.
6
5
4
H ealthy
M CI
D AT
3
2
1
0
Cor si
VS M
Results: Performance on both measures of visuo-spatial working memory
was significantly affected by dementia status. Post hoc comparisons showed
that individuals with early-stage DAT had significantly lower spans on both
the Corsi and the VSM compared to their healthy peers and to those with
MCI (see figure). However, impairment in visuo-spatial working memory in
individuals with MCI was not uniform. Their span on the VSM was comparable
to that of the healthy group whereas their span on the Corsi was significantly
lower.
Key References:
Discussion: In individuals with DS and early-stage DAT, both the visual and
spatial components of visuo-spatial working memory were impaired. In
contrast, individuals with MCI showed impairment in their spatial span but
relatively preserved visual span. Deficits in the spatial component of visuospatial working memory then, may be characteristic of DAT several years
before clinical diagnosis. These results support the view that different neural
substrates are involved in the visual and spatial aspects of visuo-spatial
working memory. In MCI, the dorsal cortical stream (responsible for processing
spatial characteristics of stimuli) may be more compromised relative to the
ventral cortical stream (responsible for processing visual characteristics) and
hence the relative sparing of function subserved by this region (1).
1. Atkinson, A., King, J., Braddick, O., Nokes, L., Anker, S., & Braddick, F. (1997). Neuroreport, 8, 1919-22
2. Grossi, D., Becker, J.T., Smith, C., Trojano, L. (1993). Psychological Medicine, 23, 65-70.
3. Kirk, S.A.,McCarthy,J.J., Kirk,W.D. (1968). ITPA-Revised. Urbana, IL: University of Illinois Press.
4. Lanfranchi, S., Cornoldi, C., & Vianello, R. (2004). American Journal of Mental Retardation, 109(6), 456-66.
5. Logie, R. (1986). The Quarterly Journal of Experimental Psychology, 38A, 229-47.
6. Milner,B. (1971). British Medical Bulletin, 3, 272-7.
This work was supported by funds from the New York State Office of Mental Retardation and Developmental
Disabilities and NIH grants R01 AG14771 (Darlynne A. Devenny) and P01 HD35897 (Wayne Silverman).
85
SYMPOSIUM 7
Neuropathological Evidence of Alzheimer’s Disease in Down Syndrome
Nelson, L.D., Kepe, V., Siddarth, P., Barrio, J., Small, G.
University of California, Los Angeles (UCLA)
Contact: [email protected]
Introduction: Neuropathological studies have shown that virtually all adults with Down syndrome (DS)
develop the neuropathology associated with Alzheimer’s disease (AD) beginning in their 3rd decade of life.
Until the recent development of neuro-imaging techniques, however, it has not been possible to show a direct
association between performance on neuropsychological measures and the presence of neuropathological
markers of AD.
Method: Participants with full trisomy 21 were examined based on neuropsychological measures and a
radio-labeled probe ([18F]FDDNP PET) that determines level and location of β-amyloid (a hallmark sign of
AD). The current study was designed to provide preliminary results from N=11 subjects with DS, ranging in
age from 23 to 50 years. Performance was assessed on a neuropsychological test battery. The unique
aspect of the test battery was that it included translational research in which five tests derived from animal
models of learning and memory were used, as well as standardized direct and indirect measures of
neuropsychological and emotional functioning. Subjects with DS were compared to age-matched healthy
controls and people diagnosed with AD from the general population.
Results: Eight PET scans were of sufficient quality to perform the analysis. Closer examination of the four
subjects with Down syndrome who demonstrated highest [18F]FDDNP values showed their ages as 30, 32,
41, and 50, consistent with the literature which supports a significant increase in neuropathology following
age 30. Of interest is that the 41 year-old and the 50 year-old were suspected of having dementia based on
their performance on the neuropsychological test battery. The 32 year-old individual performed well below
the sample mean on the measure of executive (frontal) functioning and was similarly judged as impaired by
her caregivers on frontal measures of emotional and pragmatic language functioning.
Fig 1. Representative parametric images of [18F]FDDNP.
In Fig. 1 (left), patterns of β-amyloid and neurofibrillary tangle deposition are illustrated in four subjects: A
normal control, a young person with DS, an older individual with DS, and a person with AD from the general
population. As seen here, in vivo visualization of the deposition pattern in a 41-year old individual with DS
was similar to that of an individual with AD. In the younger subject, β-amyloid and neurofibrillary tangle
deposition was similar to a control subject, with some indication of increased deposition in the posterior
cingulate gyrus.
Discussion: When related to results of functional brain imaging and neuropathology of AD, specific
neurobehavioral functions emerged which suggested early frontal involvement in the course of AD. Preliminary
results support feasibility of these methods as a means of early detection of AD in people with DS.
This work was supported by funds from the Charles Epstein award of the National Down Syndrome Society
to LDN.
Fig 1. Representative parametric images of [18F]FDDNP.
86
Symposium 8
S Y M P O S I U M
8
Behavioral Phenotypes
Chair:
Travis Thompson,
Department of Pediatrics Autism Program,
University of Minnesota School of Medicine
87
SYMPOSIUM 8
Behavioral Phenotypes
Chair: :
Travis Thompson, Department of Pediatrics Autism Program, University
of Minnesota School of Medicine
Prader-Willi Syndrome, Oxytocin, and Behavior
Elisabeth Dykens, Elizabeth Roof, Elizabeth Pantino, Rebecca Kossler, Ray Johnson, Mayim
Bialik, Hossein Nazarloo, and Sue Carter
Vanderbilt Kennedy Center
Autism: Predictors of Response to Treatment as Behavioral Phenotypes
Travis Thompson1
Glen Sallows2
1
2
Dept of Pediatrics Autism Program, University of Minnesota School of Medicine
Wisconsin Early Autism Project
Behavioral Phenotype in Relationship to Analysis of Behavioral Function and Intervention
William J. McIlvane, Curtis K, Deutsch, Krista M. Wilkinson, & William V. Dube
University of Massachusetts Medical School - Shriver Center
88
SYMPOSIUM 8
Prader-Willi Syndrome, Oxytocin, and Behavior
Elisabeth Dykens, Elizabeth Roof, Elizabeth Pantino, Rebecca Kossler, Ray Johnson, Mayim Bialik,
Hossein Nazarloo, and Sue Carter
Vanderbilt Kennedy Center, 230 Appleton Place, Nashville TN 37203
Contact: [email protected]
Introduction: Prader-Willi syndrome (PWS) is a genetic disorder associated with a distinctive behavioral
phenotype, including intellectual disabilities, compulsive behaviors, skin-picking, hyperphagia, and risks of
morbid obesity. Many persons with PWS also have increased interests in the caretaking of others,
including pets and children. Oxytocin (OT) is a neuropeptide strongly implicated in social learning and
memory, as well as in attachment, appetite regulation, and repetitive behaviors. Previous studies
identified decreased OT-secreting neurons in the hypothalamic paraventricular nuclei of adults with PWS,
as well as elevated CSF levels of OT. Aberrant OT has thus been implicated in the broad PWS
phenotype, but direct links have not yet been made between OT and specific behavioral features of PWS.
This study compared levels of OT in PWS and typical controls, and also related levels of OT and a related
peptide, vasopressin (VP), to salient aspects of the PWS behavioral phenotype.
Methods: OT and VP assays were conducted on plasma samples from 25 persons with PWS (11 males,
14 females) aged 13 to 45 years (M = 21 years). Of these, 11 had paternal deletions of chromosome 15
(q11-q13), 9 had maternal uniparental disomy, and 5 had either microdeletions or translocations.
Participants were part of a larger longitudinal study on PWS, and they and their families were individually
administered measures that tapped: hyperphagia, compulsivity, social skills, nurturance, personality, and
maladaptive behavior (e.g., Yale-Brown Obsessive-Compulsive Scale; Reiss Personality and Motivation;
Vineland Adaptive Behavior Scales; Hyperphagia Questionnaire). Levels of OT and VP were also obtained
from the plasma of 31 typical controls, matched on age, gender, and degree of obesity (BMI).
Results: Levels of OT and VP were significantly elevated in PWS relative to controls (M’s = 337.63 pg/ml
versus 144.33 pg/ml, p < .05). Within the PWS group, persons with maternal UPD had significantly lower
levels of VP compared to those with paternal deletions (50.74 versus 107.16); there was a trend for OT to
be lower in the UPD group as well. Males had significantly higher levels of OT and VP, but were otherwise
similar to females in BMI, age and behavioral measures. Levels of OT or VP were not associated with
BMI or hyperphagic behavior. VP was positively correlated with poor frustration tolerance, and with
repetitive acts and counting compulsions on the Y-BOCS. OT was positively correlated with two variants
of skin-picking in PWS: hair pulling and rectal digging, as well as with increased scores on the Vineland
Socialization scale.
Discussion: Although preliminary, findings suggest more robust ties between OT, VP and repetitive
behaviors and socialization skills than to hyperphagia. Findings will be discussed in relation to aberrant OT
in other developmental or psychiatric disorders, eg., autism and OCD. Consistent with the theme of this
symposium, implications will also be made for possible endophenotypes within PWS.
This research was supported by grants from NICHD; R0135681-07 and P30HD15052, the Vanderbilt
Kennedy Center, and the Vanderbilt GCRC.
89
SYMPOSIUM 8
Autism: Predictors of Response to Treatment as Behavioral Phenotypes
Travis Thompson and Glen Sallows
Contact:Travis Thompson, Dept of Pediatrics Autism Program, University of Minnesota School of
Medicine, Minneapolis,MN 55455 [email protected] ; Glen Sallows, Wisconsin Early Autism Project,
6402 Odana Road, Madison, WI 53719 [email protected]; Tammy L. Graupner, Wisconsin Early
Autism Project, 6402 Odana Road, Madison, WI 53719 [email protected]
Introduction: Autism Spectrum Disorders are a complex set of developmental disabilities that have
defied empirically valid sub-phenotyping. The ability to predict response to treatment is a widely used
strategy for identifying subtypes in other fields which can subsequently lead to identifying underlying
mechanisms responsible for differences. In the present report we describe early intervention outcomes for
two groups of children with autism spectrum diagnoses who received intensive early behavioral
intervention;
Methods: Children with autism (2-4 years old at enrollment) were evaluated using standard diagnostic and
psychological assessment batteries before participating in an intensive early behavior therapy program. In
one group (Wisconsin) 24 children participated in early intervention for 4 years with a mean number of 3139 hours per week. In a second group (Minnesota) children received an average of 18 hours per week
over 18 months. In the Minnesota sample 45 children participated in intensive early behavior therapy an
average of 18 hours per week. Post therapy gains were assessed at 4 years (Wisconsin) and 12-18
months(Minnesota).
Results: IQ (FS, V, P), receptive language, and ADI-R social and communication scores were significantly
associated with rapid learning in the Wisconsin sample. Children in the Rapid Learning Group had
significantly higher scores on the Early Learning Measure, a simple discrimination learning task. Children
who fell into the “rapid learner” Wisconsin group achieved most of their gains in the first 12-24 months of
early intervention. In the Minnesota sample, fifty three percent functioned sufficiently well after 18 months
to be placed in regular education classrooms with pull-out and minimal supports. 25% were in special
education classrooms for children with autism or intellectual disabilities. 4% were in regular and special
education classrooms part of the day. The remainder were home schooled. Chlldren who ended up in
regular education classrooms exhibited the most rapid and largest gains on the receptive and expressive
communication scales of the Assessment of Basic Learning and Language Scales.
Discussion: Children with autism who learn rapidly in intensive early intervention appear to correspond to
Miles et. al. (2005) Essential Autism, a familial form with a significantly higher recurrence rate within
families. Children with Miles et. al. (2005) Complex Autism do not have greater risk of recurrence within
families and appear to have experienced an early morphogenesis error, manifested by significant
dysmorphology or microcephaly. Individuals with complex autism have lower IQs (P=0.006), more seizures
(P=0.0008), more abnormal EEGs (46% vs. 30%), more brain abnormalities by MRI (28% vs. 13%), and
perform less well in interventions. Future research using gene microarrays may make it possible to further
refine Essential Autism endophenotypes and to devise targeted early interventions for children with autism
subtypes.
Miles et.al. (2005) Am J Med Genet A. 135(2):171-80.
Sallows G and Graupner, TL (2005) Am J Ment Retard 110:417-438
90
SYMPOSIUM 8
Behavioral Phenotype in Relationship to Analysis of Behavioral Function and Intervention
William J. McIlvane, Curtis K, Deutsch, Krista M. Wilkinson, & William V. Dube
University of Massachusetts Medical School - Shriver Center, 200 Trapelo Road, Waltham, MA 02452
Contact: [email protected]
Introduction: term “behavioral phenotype,” is applied to specify distinctive characteristics in subtypes of
MRDD as defined by etiology or phenomenology. Behavioral phenotypes are of interest, because taking
characteristic group differences into account may facilitate efforts to resolve population heterogeneity,
clarify causal pathways, and define clinically important targets for therapeutic interventions. Moreover, if
one is interested in a comprehensive analysis of behavioral function, it is important to know about the
degree to which behavioral phenotypes interact with such analysis and associated intervention
procedures. As the behavioral science of MRDD moves forward within a context of substantial and
growing interest in genetic and other biological processes that influence behavior, we think it important to
consider thoughtfully and to discuss the implications of the following questions:
Methods: Several programs of research will address the following issues:
1) Selected data from recent studies of children with autism and Down syndrome will examine the extent
to which research has adequately consider behavioral phenotype when developing procedures aimed at
the improvement of behavioral function.
Given that research has demonstrated statistically significant differences among different etiological
subgroups, we will consider how these differences may be considered in designing intervention
procedures aimed at prevention or amelioration of intellectual disability and associated behavioral deficits.
We will consider and evaluate data and approaches to data interpretation that suggest the need for (or
not) special procedures for responding to the characteristics of the behavioral phenotype.
2) To what extent do behavioral characteristics of individuals with Asperger syndrome inform us about
behavioral processes in children with more substantial intellectual disabilities? Research has not
adequately demonstrated that behavioral phenotypes defined for high-functioning individuals within a
given diagnostic category predict the performance of individuals whose development is less advanced?
3) Studies will be presented indicating that behavioral tendencies associated with specific diagnostic
categories are not static entities exerting ongoing influence independent of environment-behavior
interactions? Evidence will be presented from investigations of “stimulus overselectivity”, i.e. maladaptive
restricted stimulus control in learning situations – an outcome that may be especially prominent in autism
spectrum disorders. We have had substantial success in broadening stimulus control under some
circumstances, raising the questions of what the behavioral phenotype associated with this disorder
actually is and whether inferences drawn from limited studies are justifiably extended.
Discussion: Greater attention to behavioral phenotypes should be considered in developing interventions
of specific disabilities. Findings based on data from higher functioning individuals cannot be assumed to
be generalizeable to lower-functioning individuals with the same or similar disabilities. Phenotypic features
commonly associated with specific disabilities may be malleable with appropriate interventions, suggesting
they may not be immutable phenotypic characteristics.
Supported by NICHD grants HD04147, HD25995, & HD04666
91
Symposium 9
S Y M P O S I U M
9
Biobehavioral Analyses of Self-Injurious Behavior
Chair:
92
Frank J. Symons
Department of Educational Psychology
University of Minnesota
SYMPOSIUM 9
Biobehavioral Analyses of Self-Injurious Behavior
Chair: Frank J. Symons
Department of Educational Psychology
University of Minnesota, Minneapolis, MN 55455
A Comparison of the Frequency, Location, and Function of Self-Injurious Behavior between
High and Low Intensity SIB Cases
Chin-Chih Chen
University of Minnesota
A Comparison of the Frequency of Nonverbal Behavioral Indices of Pain Expression
between SIB and Control Cases During Censory Testing
Stacy Danov
University of Minnesota
Integrating Autonomic Arousal into Functional Behavioral Analysis of Self-Injury
John Hoch
University of Minnesota
A Comparison of Epidermal Morphology and Physiology between SIB and Control Cases
Frank Symons
University of Minnesota
The conceptual model for this symposium begins by recognizing that chronic SIB typically involves
repeated, forceful stimulation to specific, localized body sites (e.g. hitting specific spot on head,
picking an area on the leg, biting a specific part of the hand), rather than random patterns of SIB
distributed to various points on the body. A subset of individuals with chronic SIB and
developmental disabilities may have dysregulated pain capacities manifest in the form of altered
pain sensory thresholds and/or impairments in pain expression and regulation. This
conceptualization of SIB does not directly address etiology nor address whether pain is a cause or
consequence of SIB, but it does provide a framework to test several predictions specific to
behavioral and biological endpoints relevant to both SIB and pain. Pain mechanisms can be
indexed in a variety of ways including the properties of the stimulus (frequency, intensity, body
location), sensory reactivity and behavioral expression, autonomic response, and
neurophysiology. SIB can also be influenced by the environment, such that any examination of
physiological factors must also take into account the possibility of environmental mediation.
Ultimately, if pain mechanisms play a part some forms of self-injury, then new directions for
assessment and treatment need to be developed. In this symposium, four papers will be
presented from ongoing studies providing initial tests examining SIB along a number of biobehavioral dimensions including (a) the relation between environmental mediation and behavioral
pain parameters (frequency/body sites/intensity), (b) sensory reactivity and pain expression (c)
autonomic reactivity, and (d) neurophysiology.
93
SYMPOSIUM 9
A Comparison of the Frequency, Location, and Function of Self-Injurious Behaviors
between High and Low Intensity SIB Cases
Chin-Chih Chen1, Jamie Clary3, Dawn Parker3, Vicki Harper3,
James W. Bodfish2,3, & Frank J. Symons1
1
University of Minnesota, Minneapolis, MN 55455
2
UNC-Human Development Research Institute, 3Riddle Developmental Center
Introduction: There may be SIB subtypes that are related to the topographical or structural features of
SIB expression (frequency, location, intensity) which share a common underlying pathophysiology relevant
for biobehavioral analyses of SIB. In previous work, our group has quantified SIB location to test group
differences in syndrome specific and nonspecific developmental disabilities. In this study we tested the
hypothesis that there would be differences between high and low intensity SIB in relation to structural and
functional features of SIB. Specifically, structural (frequency, duration, location) and functional (contingent
staff interaction) features of SIB were measured and compared between matched high- and low-intensity
SIB cases. We hypothesized that high but not low intensity SIB cases would have more frequent (rate)
and more localized (% of body surface area) SIB and be less socially mediated (lower Yule’s Q values).
Methods: SIB was directly observed for three 15 min sessions for 69 individuals with chronic SIB (50%
male; mean age = 43; > 90% profound cognitive impairment). Observational data were collected during
similar contexts for each participant associated with individual support plans and rehabilitation activities.
Data were collected using hand-held iPAQ Pocket PC handheld devices with Observer 5.0 Mobile software
and were analyzed using the Observer and MOOSES observational software packages. Summary
(frequency, duration) and sequential analyses (sequential dependencies) were performed to test
differences between high and low intensity SIB cases. High (HI) and low intensity (LI) groups were formed
by scores on the Self-Injury Trauma Scale (SITS) for SIB severity index (p < .001) and number of wounds
(p < .001). Correlations were examined between the degree of environmental mediation (Yule’s Q) and
SIB frequency, duration, and location.
Results: Interobserver agreement (A/A&D) was calculated for 28% of sessions and was 93% for
frequency codes and 85% for duration codes. Overall frequency of SIB between high (0.28/min) and low
(0.32) intensity SIB groups was similar, but the mean duration of SIB was considerably greater for the HI
(302 sec) versus LI (198 sec) group (p < .06). The HI group’s SIB was more localized than the LI group
(99 vs 125 cm2). There were significant (p < .05) inverse linear relations between location (i.e. total body
surface area; TBSA) and social mediation (i.e., probability of contingent staff interaction) between the HI (r
= - 0.26) and the LI (r = 0.36) SIB subgroups. The more TBSA the less likely staff interacted contingently
to SIB for the HI group but the more likely staff interacted contingently to SIB for the LI group.
Discussion: Overall, the HI SIB subgroup spent more time in SIB directed to more localized body areas
compared with the LI group. The sequential pattern of staff interaction contingent on SIB was different
between HI and LI subgroups. Although preliminary, this set of analyses provides initial evidence for the
relevance of a structural subtype distinction based on the intensity and location of SIB
Supported, in part, by NICHD Grant No. 44763
94
SYMPOSIUM 9
A Comparison of the Frequency of Nonverbal Behavioral Indices of Pain Expression
between SIB and Control Cases during Sensory Testing
Stacy Danov1, Satomi Shinde1, Chin-Chih Chen1, Jamie Clary3, Vicki Harper3, Dawn Parker3,
James W. Bodfish2,3, & Frank J. Symons1
1
University of Minnesota, Minneapolis, MN 55455
2
UNC-Human Development Research Institute, 3Riddle Developmental Center
Introduction: There may be subtypes of SIB cases associated with altered pain experience and
expression. Because pain is difficult to assess in individuals with developmental disabilities its relation to
SIB is not well understood. This study draws upon the large body of empirical evidence for objective,
nonverbal signs of emotional expression based on changes in facial action units (FAUs; Ekman & Friesen,
1978). FAUs are anatomically based configurations of facial musculature that can be directly observed and
reliably coded and have been used to identify pain based on empirically validated facial coding of
expression during known painful events such as vaccination, blood draw, or minor invasive procedures.
The specific purpose of this preliminary study was to code and compare FAUs between a sample of adults
with chronic SIB and matched controls during a modified quantitative sensory testing protocol (mQST) to
evaluate their sensitivity and specificity.
Methods: Facial actions were examined between a chronic SIB (n = 36; 40% male; mean age = 45) and
matched control group (n = 21; 57% male; mean age = 44) during a mQST procedure. Each participant
was seated on a chair in front of a medical examining screen. A trained clinician applied five different
sensory stimulus modalities (i.e., pin prick, heat, cold, deep pressure, light touch) to the participant’s back
in a randomized order. Each participant received 10 trials that included exposure to all sensory modalities.
To ensure coders were blind, half of the trials were sham (i.e., no stimulus was applied) although the
clinician continued to signal each trial in a standardized way. A camera set up orthogonal to the participant
and focused on the face was used to collect digital video for subsequent coding using Pro-Coder Digital
Version (PCDV). Sixteen FAUs were selected for initial analyses based on reviewing the pain expression
literature with other vulnerable populations. Mean between-group differences in FAUs were examined in
addition to stimulus vs sham comparisons. A set of head movements were also measured to evaluate any
differences in head movement associated with the mQST.
Results: Inter-observer agreement was calculated on 32% of sessions and was 74% (FAUs) and 90%
(head movement). There were significant (p < .01) differences between the SIB (mean FAU freq = 1.75)
and no SIB (mean FAU freq = 0.84) control group for overall FAU activity. When FAUs were examined in
relation to modality, the SIB group had greater FAU activity for each specific modality than controls. There
were no between group differences for head movement (turn, back, forward). Mean FAU activity during
stimulus trials was greater than during sham trials although this difference was not significant.
Discussion: Overall there were differences between SIB and matched controls in FAU activity
(sensitivity). Although preliminary, the observation that there were different FAU activity patterns is
consistent with the possibility of altered sensory expression associated with chronic SIB but a more
specific measure may be necessary.
Supported, in part, by NICHD Grant No. 44763
95
SYMPOSIUM 9
Integrating Autonomic Arousal into Functional Behavioral Analysis of Self-Injury
John Hoch1, Mika Tarvainen2, & Frank J. Symons1,
University of Minnesota; 2University of Kuopio, Finland
1
Introduction: Little is known about the role of internal physiological processes underlying severe problem
behaviors. By integrating real time observation of behavior with online recording of autonomic function,
researchers could gain insight into problem behavior that is unresponsive to social contingencies. This
study extends previous work by Freeman et al, (1999) by recording RR interval heart rate data allowing
more precise quantification of heart rate variability as well as collecting the data during classroom based
functional behavioral analyses (FBA) allowing for greater experimental control over consequences for
problem behavior.
Methods: The relative influence of problem behavior and changes in FBA condition on derived cardiac
measures (heart rate, heart rate variability and sample entropy) were analyzed using time lag sequential
analysis for three children with developmental disabilities and self injury and aggression. Yule’s Q’s of
changes in cardiac measures before and after problem behavior and changes in FBA conditions were
compared using a resampling procedure (Yoder, et al 2001). Within-subject ANOVA analyses were also
employed to examine the variance in cardiac measures accounted for by problem behavior and FBA
conditions.
Results: Correlations were higher for FBA condition than for problem behavior for all participants despite
the lack of differentiation of problem behavior across conditions. Sequential analysis found no changes in
heart rate greater than 1 standard deviation than the participant’s baseline values in the 10 seconds
preceding or following problem behavior compared to chance occurrence for the participants. No
sequential associations were found preceding or following changes in FBA conditions.
Discussion: This study suggests that integrative analysis of physiological arousal can be used to discern
the role of autonomic arousal in self injury or other forms of destructive behavior. Documenting autonomic
changes may be relevant to discerning possible subtypes. The methodology demonstrated here improves
our understanding of behavior that is not differentiated in FBA’s by examining variation in underlying
physiological processes. Limitations include the self selected nature of the sample and missing heart rate
data due to participant refusal to wear device.
Supported, in part, by NICHD Grant No. 44763
96
SYMPOSIUM 9
A Comparison of Epidermal Morphology and Physiology between SIB and Control Cases
Frank J. Symons1, Gwen Wendelschafer-Crabb1, Elizabeth Gilles2,
William Kennedy1, & James W. Bodfish, 3,4
1
University of Minnesota, Minneapolis, MN 55455
2
Gillette Children’s Specialty Healthcare
3
UNC-Human Development Research Institute, 4Riddle Developmental Center
Introduction: We hypothezised that there may be a SIB subtype associated with a specific pattern of
morphological and neurochemical markers characterized by altered function or structure of the sensory
nerve fibers involved in pain signaling and by altered concentrations of neuropeptides involved in pain and
stress signaling. The purpose of this preliminary study was to examine the morphology and neuropeptide
density of epidermal nerve fibers quantified through skin biopsy samples from individuals with
neurodevelopmental disorders and chronic SIB compared with a non-SIB normal and MA-matched
controls.
Methods: A cross-sectional design was used with 3 mm punch skin biopsies collected from each
participant from non-self-injurious body sites and compared with site-matched existing normal control skin
samples. The study was conducted at an adult and pediatric outpatient clinic and tertiary care facility.
Twenty-five adults (mean age = 41; 65% male; 80% profound cognitive impairment) and 5 children (mean
age = 12; 3 male; 100 % profound cognitive impairment) with severe SIB secondary to intellectual and
developmental disability were recruited. The primary dependent measure for the morphology analyses was
the coefficient of variation (CV) to quantify the mean gap length between epidermal nerve fibers for each
subject.
Results: Full quantitative analyses have been completed for 10 participants. Visual microscopic
examination and qualitative analyses of the microscopy images suggest there are morphological
abnormalities characterized as ‘sprouting’ or ‘tufting’ abnormalities in the epidermal nerve fibers among a
subset of chronic SIB cases (similar in some case with that observed in small diameter peripheral
neuropathies). In 50% of the cases analyzed to date with all fibers counted, variations in ENF distribution
resulting from the tufting and clustering of nerve fibers in the SIB cases were clearly observed. Analysis of
peptides showed normal levels of CGRP but SP levels were elevated in the chronic SIB cases. Substance
P (SP) fiber density was increased with 2 to 3 times as many fibers in SIB subjects as normal IQ control
participants and between SIB and developmental disability control participants.
Discussion: We found preliminary evidence for altered morphology (i.e., tightly clustered large groupings
of nerves with relatively long intervening spaces) and neurochemistry (i.e., elevated SP fiber density).
These results from this novel immunohistological analysis of skin in individuals with severe neurological
impairment and SIB do not definitively confirm or refute a subtype model, but do suggest that there may
be significant differences in the morphology of nerve fibers in at least a portion of chronic SIB cases. The
significance of these findings is underscored by the fact that the biopsy was sampled at a non-SIB site
with no history of tissue damage.
Supported, in part, by NICHD Grant No. 44763
97
Pa p e r Se s s i o n 1
P A P E R
1
FRAGILE X AND AUTISM
98
PAPER SESSION 1
Educational & Therapeutic Involvement by Parents of Young Children with ASD
Paul R. Benson
Center for Social Development & Education, University of Massachusetts, Boston
100 Morrissey Blvd., Boston MA 02125
Contact: [email protected]
The importance of parent involvement in educational & therapeutic interventions for young children with
autism spectrum disorders (ASD) is well documented. However, despite its importance, relatively little
research has been conducted on this topic. To address this issue, we implemented a longitudinal
research project which collected data on parent educational & therapeutic involvement, child & family
functioning, & school & non-school services. Questionnaire & interview data from 98 parents of young
children with ASD were gathered at 3 points in time approximately a year apart. In addition, teacher data
on child classroom behavior & functioning were also gathered annually using standardized assessment
instruments. Utilizing these data, & guided by a conceptual model drawing upon ecocultural theory &
studies of family adaptation to disability, we examined 5 types of parent involvement: (1) school-based
educational involvement; (2) home-based educational involvement; (3) parent facilitation of professional
therapies outside of school; (4) parent facilitation of biomedical interventions; & (5) parent educational
advocacy.
Our presentation of preliminary study findings will focus on a discussion of changes in parent involvement
over time as well as multivariate analyses predicting intensity of parent school- & home-based involvement
at T1 & T3.
Change in involvement intensity for the 5 involvement types were assessed at both the group- &
individual-level. Group-level change in intensity of parent involvement was found to be relatively stable
over the study’s three time points, however, substantial individual-level change was also observed, with
the greatest individual change occurring in parent home-based involvement & the least in parent
facilitation of biomedical interventions.
Separate regression analyses were used to assess the impact of child, school, & family factors on
intensity of parent school- & home-based involvement. Significant positive predictors of T1 school
involvement included parent developmental expectations, parent social support, & school facilitation of
parent school involvement. T1 home involvement was significantly predicted by family SES, child adaptive
functioning, childcare demands, & school facilitation of parent home-based involvement. In both T1
regressions, school facilitation was found, by far, to be the most powerful factor affecting intensity of
parent involvement at home & at school.
Regression analyses were also used to assess the impact of change in child, school, & family factors on
change in school- & home-based involvement. As in the previous T1 regressions, across these T1-T3
analyses, school facilitation was found to be the single most powerful predictor of change in involvement,
with increases in school facilitation being positively linked to increases in parent involvement between T1
and T3. In addition, consistent with the tenets of ecocultural theory, a measure of meaningfulness (i.e.,
the extent to which parents reported involvement activities to be consistent with core family values &
goals) was found to significantly predict intensity of both parent school- & home-based involvement, with
higher meaningfulness scores at Time 1 & an increase in meaningfulness scores between Time 1 & Time
3 being associated with higher levels of parent school- & home-based educational involvement at Time 3.
Implications of study findings for future research & for the support of parents seeking to participate in the
education and treatment of their children with ASD will be discussed.
99
PAPER SESSION 1
Neural Network Analysis of MAOA, Serotonin Transporter, and Serotonin Receptor (HTR1B)
Polymorphisms on the Behaviors of Children on the Autism Spectrum
Cohen, I.L.*1, Liu, X.2, Jenkins, E.C.1, Brown, W.T. 1, Gonzalez, M. 1, Rovito-Gomez, T. 1, Kim, S-Y1,
Lewis, S. 2, Chudley, A. 2, ASD-CARC, and Holden, J.J.A.2
1
NYS Institute for Basic Research in Developmental Disabilities, Staten Island, NY, 10314
2
Queens University, Kingston, Ontario, K7M, 8A6
Introduction: We found that autistic behaviors are correlated with an interaction between a functional
polymorphism in the MAOA promoter region and the functional serotonin transporter gene promoter
polymorphism (5-HTTLPR) (Holden, et al., 2006). These data suggested a need to take into account gene
interactions. Neural network models are useful for this purpose. They were employed to examine the
additional contribution of the G861C SNP in the HTR1B gene to the above-described associations in boys
and girls on the autism spectrum (n=245 to 274). The HTR1B receptor is important in regulating the
release of serotonin into the synapse and the C allele of this SNP has been found to be more common in
affected children and to be associated with more severe symptoms (Liu, et al., 2002).
Method: The low or high activity allele status of all cases followed that suggested by Deckert, et al.
(1999). MAOA analysis was done as described previously (Cohen, et al., 2004). 5-HTTLPR alleles were
characterized as short or long. HTR1B alleles were G or C. Behavior patterns were assessed with the
PDD Behavior Inventory (PDDBI; Cohen and Sudhalter, 2005). Back-propagation models predicted
behavior scores based on various allelic combinations, with age, sex, presence of autism in a sibling, and
data source (Canada or USA) serving as control variables. K-fold cross-validation (K=10) was employed.
Results: Mean (SE) Pearson correlations for the training, and validation sets varied across allelic
combinations. The strongest correlations in the validation set were for domains assessing receptive and
expressive language competence.
Discussion: Neural network models may prove a fruitful approach in identifying genotype-phenotype
correlations in this population.
Key References:
Cohen IL et al (2003) Association of autism severity with a monoamine oxidase A functional polymorphism.
Clin Genet 64:190-197
Cohen IL, Sudhalter V. 2005. The PDD Behavior Inventory. Lutz, FL: Psychological Assessment
Resources, Inc.
Deckert J et al (1999) Excess of high activity monoamine oxidase A gene promoter alleles in female
patients with panic disorder. Hum.Mol.Genet 8 (4):621-624.
Holden, J.J.A., Liu, X., Flory, M., Jenkins, E.C., Brown, W.T., Lewis, S., Chudley, A., ASD-CARC,
Gonzalez, M., Rovito-Gomez, T., and Cohen, I.L. (2006). Effects of maternal and child MAOA and
serotonin transporter polymorphisms on the behavior patterns of children on the autism spectrum. Poster
Presentation, International Meeting for Autism Research, Montreal.
Li W, Haghighi F and Falk CT (1999) Design of artificial neural network and its applications to the analysis
of alcoholism data. Genetic Epidemiology 17 Suppl 1:S223-S228.
Source of Funding: March of Dimes, Canadian Institutes of Health Research, Ontario Mental Health
Foundation.
100
PAPER SESSION 1
Insomnia in Children with Autism Spectrum Disorders: Evidence from Multiple Sources
Lynnette M. Henderson, Beth A. Malow, Mary L. Marzec, Susan G. McGrew, Lily Wang, Wendy L. Stone
Introduction: Insomnia, defined as difficulty initiating or maintaining sleep, has been documented
consistently in children with autism spectrum disorders in studies using parentally completed
questionnaires, and sleep diaries1. While parental sleep concerns have been well documented in children
with ASD, few studies have related these concerns to polysomnographic (PSG) findings. Documenting
PSG abnormalities in children with ASD whose parents report sleep concerns provides objective evidence
of disordered sleep in these children.
Methods: Four to ten year-old children who were free of psychotropic medications, with no history of
mental retardation or epileptic seizures participated in 2 nights of PSG recordings. The sample included 21
children with ASD, defined as “good sleepers” (10 children) and “poor sleepers” (11 children) on the basis
of parental report and 10 typically developing (TD) children reported by their parents to be good sleepers.
Parents described their children through the Child Sleep Habits Questionnaire (CSHQ) and Child Behavior
Checklist (CBCL). Research observations included the Peabody Picture Vocabulary Test (PPVT) and the
Autism Diagnostic Observation Schedule (ADOS).
Results: Poor sleepers with ASD showed prolonged sleep latency and decreased sleep efficiency on night
1 of PSG and differed on insomnia-related subscales of the CSHQ; (increased sleep onset delay and
decreased sleep duration). The good sleepers with ASD did not differ from the TD children in sleep
architecture or on CSHQ domains. As compared with ASD good sleepers, the ASD poor sleepers also had
higher scores related to affective problems on the CBCL and more problems with reciprocal social
interaction on the ADOS. The communication (A) items and total (A + B) ADOS scores did not differ
between ASD good and poor sleepers.
Discussion: Our work demonstrates that some children with ASD sleep well and exhibited similar sleep
patterns to age-comparable TD children. In summary, our findings support that children with ASD and
parental sleep concerns differ from those without parental sleep concerns on PSG measures, sleep
duration and sleep onset delay, affective problems, and social interaction. Unlike the CBCL findings, in
which the response may have been influenced by parental perception of sleep, the ADOS was given by
examiners not aware of the child’s classification into the ASD good or poor sleeper groups, or results of
the PSG studies. The direction or causality of these relationships can not be determined from these
results, but must be examined in future studies. Interventional studies aimed at improving sleep through
pharmacological therapies, behavioral treatments, or both will be necessary to determine if improving
sleep in children with ASD can positively influence daytime behavior and autism symptomatology2.
Key References:
1.
Couturier JL, Speechley KN, Steele M, Norman R, Stringer B, Nicolson R. Parental perception of
sleep problems in children of normal intelligence with pervasive developmental disorders:
prevalence, severity, and pattern. J Am Acad Child Adolesc Psychiatry 2005;44:815-22.
2.
Malow BA, McGrew SG, Harvey M, Henderson LM, Stone WL. Impact of treating sleep apnea in a
child with autism spectrum disorder. Pediatr Neurol 2006;34:325-8.
101
PAPER SESSION 1
Girls With Fragile X Syndrome: Early Development and Behavior
Deborah D. Hatton, John Sideris, Anne Wheeler, The University of North Carolina at Chapel Hill
Kelly Sullivan, Duke University
Contact: Deborah D. Hatton, UNC-Chapel Hill,
CB 8040, Chapel Hill, NC 27599-8040
[email protected]
The purpose of this session is to describe early development and behavior in girls with fragile X syndrome
(FXS), the leading inherited cause of intellectual disability. Although research on FXS has increased
exponentially in the past decade, very little is known about young girls with FXS. This scarcity of research
on girls may be because many are not as severely affected as boys, and they may not exhibit as many
challenging behaviors. The shyness and anxiety that seem to characterize many girls with FXS may not
disrupt classroom and family life to the same extent that challenging behaviors in boys do. In some girls
with FXS, developmental delays may be subtle and more difficult to detect than those that are evident in
boys. Therefore, our goal was to examine development and behavior in a relatively young sample of girls
with FXS to see if they do, in fact, exhibit delays and problem behaviors.
To examine early development, we used hierarchical modeling to examine mean levels and rates of
development in 16 girls with full mutation FXS between the ages of 6 months and 9 years. Using the
Battelle Developmental Inventory as our outcome measure, we found considerable variability in
development. Chronological age and autistic behavior interacted, with the developmental trajectories of
girls with more autistic behavior diverging from those of girls with less autistic behavior over time. In a
subsample of 11 girls with genetic data, we found that girls with higher levels of the fragile X mental
retardation protein whose mothers had some college education had better developmental outcome.
We examined adaptive and problem behavior in 34 girls with FXS, ages infancy to 14 years, using the
Vineland Adaptive Behavior Scales, the Child Behavior Checklist, and the Teacher Report Form.
Consistent with early development, considerable variability was evident in adaptive and problem behavior.
Girls with fewer autistic behaviors had better adaptive behavior skills. The most prevalent problem
behaviors in girls were attention problems and anxiety. Teacher and parent ratings of problem behavior in
a subsample of 13 girls with ratings from both parents and teachers showed both similarities and
differences.
Our findings suggest that many girls with FXS experience delays in development and in adaptive behavior.
In addition, many experience problem behavior based on both parent and teacher report. By being attuned
to early signs of developmental delays, internalizing problems such as anxiety, and externalizing problems
such as inattention, parents and professionals can take a proactive approach to facilitating development
and learning and providing support for potential social, emotional, and behavioral problems.
102
PAPER SESSION 1
Negative Reactivity in Preschool Boys with Fragile X Syndrome
Jane E. Roberts, Meghan Shanahan, and Deborah D. Hatton
University of North Carolina
517 South Greensboro Street,
Chapel Hill NC 27599-8040,
Contact: [email protected]
Introduction. The full mutation of fragile X syndrome (FXS) is associated with moderate intellectual
disability in affected males. Despite moderate to severe cognitive delays in males with the full mutation,
parents and professionals are often most concerned about behaviors that disrupt daily life. Age may be an
important factor contributing to the occurrence and severity of problem behavior in boys with FXS.
However, very few studies of behavior in FXS during the first five years of life have been published.
Preliminary findings using cross-sectional data and parental reports suggest that behavioral challenges of
children with FXS may be subtle or absent during the infant and toddler years yet emerge and intensify in
early childhood. Given findings that negative reactivity during the preschool years may predict significant
social, cognitive, and behavior problems later in development, studies of the predictors and correlates of
negative reactivity are critical to identify risk factors for those susceptible to these problems, as well as
recognize opportunities to provide intervention to obviate/minimize these problems. Although defined
differently by investigators, it is generally acknowledged that negative reactivity includes responses of
anger, frustration, fear, and sadness to stimuli. The purpose of this study was to examine negative
reactivity in young boys with fragile X syndrome (FXS).
Method. Two groups participated in this study: 1) 25 boys with the full mutation of FXS, and 2) 64
typically-developing boys. The FXS and comparison groups were matched on chronological age (M =
35.08 months and M = 36.25 months, respectively), race (96% and 97% Caucasian, respectively), and
maternal education levels (15.9 years and 15.7 years, respectively). Multiple measures of negative
reactivity were collected including a laboratory based measure of temperament (arm restraint episode
from the Lab-TAB) that included facial sadness, facial anger, bodily struggle, and distress vocalizations. In
addition, the anger and sadness subscales from the parent-completed Child Behavior Questionnaire were
collected. The Mullen Scales of Early Learning were administered to the group with FXS to examine the
relations of mental age to negative reactivity in FXS
Results. Boys with FXS showed less facial sadness on the Lab-TAB, and their parents rated them as
exhibiting less anger and sadness on the Child Behavior Questionnaire. No group differences were found
on the Lab-TAB measures of distress vocalizations, bodily struggle, and facial anger. However, both
groups displayed a phase effect for facial anger on the Lab-TAB. Specifically, both groups’ anger
appeared to peak half way through the arm restraint episode. A moderate positive correlation between
mental age and parent perceptions of sadness from the CBQ was evident; however, no other significant
associations were found between mental age and measures of negative reactivity in boys with FXS.
Discussion. The findings of this study provide important preliminary information regarding negative
reactivity in 3-year old boys with FXS using multiple measures. Interestingly, boys with FXS displayed
significantly less sadness than the typically developing group during the laboratory task, and their mothers
rated them as displaying less sadness and anger on the CBQ than the mothers of the children in the
typically developing group. Our finding of less anger and sadness in 3-year old boys with FXS was
unexpected due to published reports of challenging behavior in older boys with FXS and suggests age
related changes in negative reactivity in boys with FXS that should be examined longitudinally.
103
PAPER SESSION 1
Psychotropic Medication Use in Fragile X Syndrome: Analysis of Side Effects
Maria G. Valdovinos, Rahul A. Parsa, Michelle L. Alexander
Drake University, Psychology Department, 2507 University Ave, Des Moines, IA 50312
Contact: [email protected]
Introduction: Fragile X syndrome (FXS) is the most common inherited cause of mental retardation and is
associated with the lack of fragile X mental retardation protein (FMRP). The behavioral phenotype
associated with FXS includes hyperactivity, anxiety, and sometimes problem behaviors such as
aggression, self-injurious behavior, and tantrums. Stimulants, antipsychotics, SSRI antidepressants and
alpha agonists are the most commonly prescribed psychotropics used to treat the associated phenotype
(Amaria, Billeisen, & Hagerman 2001; Berry-Kravis & Potanos, 2004). Very little information is known
about the side effects individuals with FXS may experience as a result of taking these medications. The
goal of this study was to determine if there were specific side effects experienced by individuals with FXS
as a result of using specific psychotropic medications.
Method: To evaluate this relationship, a nation-wide survey was conducted of caregivers of individuals
with FXS. A questionnaire developed for this study (based on a version used by Valdovinos and
Schroeder (2003)) was mailed to participants and contained items regarding age, gender, ethnicity, coexisting health conditions, psychiatric diagnoses, level of functioning, and behavioral issues and questions
regarding current and prior psychotropic medications use, specifically, the name of medication, dose,
purpose, prescribing physician, date initiated, effectiveness, effect on target behavior, and changes in
psychotropic medication in response to side effects. With regards to side effects experienced, a list of the
most common side effects associated with psychotropic drugs, as well as those most commonly
experienced by people with FXS were provided but respondents were also given the opportunity to list
additional side effects not included in the list.
Results: We received 400 surveys out of the 3099 distributed resulting in a 12.9% response rate. In
conducting our analysis, we consider each drug-respondent combination as one sample unit. Given the
small number of respondents, we decided to group the drugs by drug class as opposed to evaluating
individual drugs. We also grouped side effects into classes. Our preliminary results indicate that atypical
antipsychotic drugs had higher than expected gastrointestinal side effects (weight gain and increase in
appetite) and fewer than expected sleep (sedation) and overall health side effects. Stimulants had higher
than expected gastrointestinal (weight loss and decrease in appetite) and neurological side effects (tics,
twitching, and strange movements) and fewer sleep side effects (sedation). Alpha-adrenergic stimulant
group had much higher than expected sleep side effects (sedation) and less than expected overall health
side effects. These data were analyzed using a chi-square test of independence, resulting in a p-value
0.0014.
Discussion: Based on our preliminary analysis, the side effects profiles were consistent with what is
reported for the general population with a few exceptions. This presentation will include the final analysis
of our data.
Key References:
Amaria, R. N., Billeisen, L. L., & Hagerman, R. (2001). Medication use in Fragile X syndrome. Mental Health
Aspects of Developmental Disabilities, 4, 143-147.
Berry-Kravis, E., & Potanos, K. (2004). Psychopharmacology in Fragile X syndrome – Present and future. Mental
Retardation and Developmental Disabilities Research Reviews, 10, 42-48.
Valdovinos, M. G., & Schroeder, S. R. (2003). The effects of a computerized monitoring system of psychotropic
medication use by people with developmental disabilities on staff reported side effects. Journal of
Developmental and Physical Disabilities, 15, 299-334.
* This research was supported by a Clinical Research Award from the National Fragile X Foundation
104
Pa p e r Se s s i o n 2
P A P E R
2
FAMILIES, MENTAL HEALTH, AND SOCIAL BEHAVIOR
105
PAPER SESSION 2
The Pivotal Behavior Model of Development:
Implications for Developmental Intervention
Gerald Mahoney Frida Perales
Mandel School of Applied Social Sciences,
Case Western Reserve University
Contact: [email protected]
Pivotal behaviors are the learning processes that enable children to acquire discrete skills that are not
directly targeted in intervention (Koegel, Koegel & Carter, 1999). The concept of pivotal behavior evolved
from applied behavioral analysis intervention research which historically has focused on discrete skill
interventions. It evolved from the concern that the process of teaching discrete targeted behaviors to
children one at a time (as incurs in programs such as Discrete Trial Training) is slow, laborious and
ineffective at promoting a broad range developmental outcomes. By teaching children to use pivotal skills,
there is some evidence that children spontaneously engage in the types of interactions that result in
learning across broad numbers of behaviors (Humphries, 2003).
In this presentation we will present data indicating that the concept of pivotal behavior may be useful both
for understanding parental influences on children’s learning and development and for developing parentmediated models of developmental intervention. Two studies of young children with disabilities and their
mothers will be described.
The first is a descriptive study of 45 mother-child dyads in which the children are less than three years old
and have significant developmental disabilities (Mahoney, Kim & Linn, In Press). Videotaped observations
of mother child interactions were used to code mothers’ style of interaction using the Maternal Behavior
Rating Scale (Mahoney, 1999) and seven of children’s pivotal behaviors using the Child Behavior Rating
Scale (Mahoney & Wheeden 1998). Analyses indicated that children’s level of pivotal behaviors was
associated both with their mothers’ level of responsiveness and with children’s rate of development as
measured by the Vineland Adaptive Behavior Scale and the Transdisciplinary Play Based Assessment.
Regression analyses suggested that children’s pivotal behavior mediated the relationship between
maternal responsiveness and children’s development.
The second is a one year intervention study that involved 50 mother-child dyads including 30 children with
developmental disabilities and a matched sample of 20 children with autism (Mahoney & Perales, 2005).
Each of these dyads received a parent-mediated intervention in which parents were encouraged to
promote children’s pivotal behaviors by using Responsive Interaction strategies in their routine interactions
with them. There were four findings from this study. First, responsive interactive strategies were effective
in encouraging parents to engage in more responsive interactions with their children. Second, there were
highly significant increases in children’s pivotal behaviors during the course of intervention. Changes in
mothers’ responsiveness accounted for 20% of the variance of the improvements in children’s pivotal
behavior. Third, children made dramatic improvements in their rate of cognitive and communication
development. In addition children with autism showed marked improvements in their social emotional
functioning as indicated by their scores on two standardized tests. Fourth, Changes in children’s pivotal
behavior accounted for 10% of the variance of the improvements in children’s rate of cognitive and
communication development.
In general, results from these two studies underscore the critical role that pivotal behavior may play in
developmental intervention. They suggest that children’s pivotal behavior plays a major role in mediating
children’s developmental learning, and that individual differences in children’s pivotal behavior are related
parental responsiveness.
106
PAPER SESSION 2
Predicting Disruptive Behavior Disorders in Five-Year Old Children
with and without Intellectual Disability
Bruce L. Baker (Dept. of Psychology, UCLA, Los Angeles, CA 90095
Rachel Fenning (Department of Psychology, UCLA)
Jan Blacher (Graduate School of Education, University of California, Riverside)
Contact: [email protected]
Introduction: Children with intellectual disability (ID) are at heightened risk for behavioral and mental
disorders. Despite notable variability in samples and assessment methods, between one third and one
half of these children have clinically significant disturbances, and at least three times as many children
with ID meet diagnostic criteria as those with typical cognitive development. This co-morbidity of any level
of ID and one or more identifiable mental disorders (APA, 2000), or dual diagnosis, is a significant burden
for the children, their families, and the social service system. Many recent studies focused on
epidemiology, but there is little known about the risk factors for mental disorder in this population. The
NICHD-supported Collaborative Family Study, at UCLA, UC Riverside, and Penn State University, has
been following children with and without developmental delays and their families from child age 3. The
present paper will examine evidence for diagnosable mental disorders as early as child age 5, consider
the odds ratio between ID and typically developing children, and consider concurrent and earlier variables
that relate to the most salient problem type: Disruptive behavior disorders.
Methods: We followed n=98 children with borderline to moderate ID and n=144 typically developing
children from child age 3 to 5 years. Overall, the child sample was 57% boys and 61% Caucasian. The
parent sample was 86% married and 51% of mothers and fathers college graduates. The SES (mothers’
education and family income) was lower in the ID group, so this was co-varied in all status group
comparisons. At child ages 3 and 5 parents completed the 1 ½ to 5 year version of the Child Behavior
Checklist (Achenbach, 2001) and, at age 5, the Diagnostic Interview Schedule for Children (DISC;
Edelbrock, Crnic & Bohnert, 1999), a highly structured diagnostic interview schedule covering current DSM
criteria for child psychiatric disorders. Measures taken at age 3 included a measure of family stress
(Family Impact Questionnaire, Donenberg & Baker, 1993) and naturalistic observations of family
interactions.
Results: At age 5, fully 58.2% of the ID sample met criteria for at least one mental disorder. The most
prevalent disorders were disruptive behavior disorders (DBD): Oppositional Defiant Disorder, 42.9% and
Attention Deficit Hyperactivity Disorder 39.8%. Separation Anxiety Disorder was the next most prevalent
(14.3%). The prevalence of every disorder was higher in the ID group. The highest difference was for
ADHD, odds ratio 3.18. Interestingly, in contrast to typically developing samples, there was no relationship
between child sex and DISC diagnoses. Child behavior disorders, parenting stress, and aspects of the
parent-child interaction at age 3 all were significantly related to DBD diagnoses two years later.
Discussion: These findings of a high rate of diagnosable disorders in children with ID as young as age 5,
have implications for broadening the focus of early intervention programs beyond cognitive abilities, to
include a focus on developing self-regulation and decreasing behavior problems.
107
PAPER SESSION 2
Mental Health of Children and Adolescents with and without
Intellectual Disabilities in Britain
Eric Emerson & Chris Hatton
Institute for Health Research, Lancaster University, UK
Contact: [email protected]
Introduction: Existing research has reported higher rates of psychopathology among children with
intellectual disabilities. There have, however, been very few studies that have investigated the prevalence
of specific psychiatric disorders (defined in accordance with DSM or ICD classification systems) and comorbidity between psychiatric disorders in well constructed population based samples of children with and
without intellectual disabilities. In addition, few studies have explored the extent to which risk factors for
psychiatric disorders (and co-morbidity) vary across children with and without intellectual disabilities.
Methods: Secondary analysis of data collected in the 1999 and 2004 Office for National Statistics (ONS)
surveys of child and adolescent mental health in Britain. These two cross sectional surveys were based on
nationally representative samples of 10,500 (1999) and 8,000 (2004) British children aged 5-15.
Psychiatric disorders were identified through well validated structured interview formats undertaken with a
primary carer (usually the child’s mother) and the child themselves (if aged 11 years or older). In previous
studies we have operationally identified children with intellectual disabilities in the 1999 data are reported
on the prevalence of psychiatric disorders and relationship between mental health and life events
(Emerson, 2003; Hatton & Emerson, 2004).
Results: In preliminary analyses we have operationally identified children with intellectual disabilities in the
2004 data and, given that there were no marked differences in overall prevalence between 1999 and
2004, merged the data files. The merged file contains approximately 500 children with intellectual
disabilities and 17,500 children without intellectual disabilities. Subsequent analyses will: (1) estimate the
prevalence of specific psychiatric disorders and combinations of co-morbid disorders among children with
and without intellectual disabilities; (2) explore the extent to which age, gender, socio-economic position,
household composition and family functioning operate as risk factors for psychiatric disorders (and comorbidity) across children with and without intellectual disabilities. The latter analyses will involve both
simple bivariate analyses and multivariate analyses (logistic regression) to identify the unique contribution
of particular factors.
Discussion: Implications for future research will be identified.
Key References
Emerson, E. (2003). The prevalence of psychiatric disorders in children and adolescents with and without
intellectual disabilities. Journal of Intellectual Disability Research 47, 51-58.
Hatton, C., & Emerson, E. (2004). The relationship between life events and psychopathology amongst
children with intellectual disabilities. Journal of Applied Research in Intellectual Disabilities 17(2), 109-118.
108
PAPER SESSION 2
Household Task Participation of Children with and without Attentional Problems
*M. Louise Dunn, ScD, OTR/L, Assistant Professor, Division of Occupational Therapy University of Utah,
520 Wakara Way, Salt Lake City, Utah 84108
Contact: [email protected]
Wendy J. Coster, PhD, OTR/L, Chair, Dept of Occupational Therapy, Boston University
Gael S. Orsmond, PhD, Assistant Prof, Dept of Occupational Therapy, Boston University
Ellen S. Cohn, ScD, OTR/L, Associate Prof, Dept of Occupational Therapy, Boston University
In this oral presentation, the first author will share information on the findings from a study examining
household participation patterns for children with and without Attention Deficit Hyperactivity Disorder
(ADHD). Children with ADHD often demonstrate deficits in their adaptive functioning including their ability
to participate in daily household routines such as household tasks (Stein et al., 1995). Engagement in
household tasks is part of being a family and may be one way that families prepare children for adult roles.
Families who have children with ADHD often expend much time and effort accommodating their children’s
needs for individualized attention by adapting their personal and family routines (Cronin, 2004; Klassen,
Miller, & Fine, 2004). Therefore, the purpose for this study was to examine household tasks participation in
school-aged children with and without ADHD because of its impact on their families and on the children’s
preparation for living in the community as an adult.
Forty-five parents of children in grades 3 through 5 (22 who had a child with ADHD and 23 whose child
had no disabilities) participated in this descriptive, cross sectional study. Results showed that the children
with ADHD did not differ significantly from children without ADHD in how many household tasks they
performed. Children with ADHD did require significantly more assistance than did their peers. Both groups
of parents reported that their children’s participation in household tasks was important. Content analysis of
parents’ reasons for involving their children in household tasks yielded three themes: personal
responsibility, social responsibility, and needing help. These findings support consideration of the time,
efforts, and costs to families in supporting the participation of their children with ADHD in home activities
when planning intervention.
After presenting my study, I would encourage discussion about learning opportunities and outcomes that
occur through participation in household routines for school-aged children with ADHD and other
disabilities. In particular, I would encourage discussion about bidirectional effects between children with
disabilities and their families on engagement in family activities.
References
Stein, M.A., Szumowski, E., Blondis, T. A., & Roizen, N. J. (1995) Adaptive skills dysfunction in
ADD and ADHD children. Journal of Child Psychology and Psychiatry. 36, 663-670.
Cronin, A. F. (2004). Mothering a child with hidden impairments. American Journal of Occupational
Therapy, 58, 83-92.
Klassen A. F, Miller, A., & Fine S. (2004). Health-related quality of life in children and adolescents
who have a diagnosis of Attention-Deficit/Hyperactivity Disorder. Pediatrics, 114, 541-547.
109
PAPER SESSION 2
Socio-Economic Position, Social Connectedness and the Self-Reported
Well-Being of Women and Men with Intellectual Disabilities in England
Chris Hatton and Eric Emerson
Institute for Health Research, Lancaster University, UK
Contact: Chris Hatton, Institute for Health Research,
Lancaster University, Lancaster, LA1 4YT, UK
[email protected]
Introduction:
Few large-scale surveys of the subjective well-being (SWB) of adults with intellectual disabilities exist, and
there is current controversy over the extent to which indicators of SWB are independent of factors such as
personal characteristics, relationships with other people and external living conditions.
Methods:
We investigated associations between five self-reported indicators of SWB and the personal
characteristics, socio-economic position and social connectedness of a sample of 1,273 English adults
with intellectual disabilities, collected as part of a national survey of adults with intellectual disabilities in
England (Emerson et al., 2005).
Results:
Mean overall life satisfaction was 81% of the scale maximum, marginally higher than typically reported
among the general population. Variation in SWB was strongly and consistently related to socioeconomic
disadvantage and, to a lesser extent, social relationships. For women, being single was associated with
greater well-being on all indicators. For men there was no association between marital status and wellbeing. Relationships with friends with intellectual disabilities appeared to be protective against feeling
helpless. For women, but not for men, increased contact with relatives was associated with greater overall
well-being.
Discussion:
The results confirm the generally high levels of SWB reported by adults with intellectual disabilities in the
face of objectively poor external living conditions, although within the sample SWB was consistently
associated with socio-economic position, suggesting that SWB is not completely independent of the
effects of socio-economic disadvantage. This study also suggests that gender effects in SWB require
more research attention.
Key References:
Emerson, E., Malam, S., Davies, I., & Spencer, K. (2005). Adults with learning difficulties in England 2003/
4.Leeds: Health & Social Care Information Centre.
110
PAPER SESSION 2
Unmet Need in Families of Young People with Intellectual and Developmental Disabilities
1
Vivien J Moffat 1 Jennifer G Wishart 2 Eve C Johnstone 1
Division of Psychiatry, 2 Moray House School of Education, University of Edinburgh
Contact: Vivien Moffat,
University of Edinburgh, Kennedy Tower,
Royal Edinburgh Hospital,
Morningside Park, Edinburgh, EH10 5HF, UK
[email protected]
Introduction: There is increasing awareness internationally of the needs of families of people with
intellectual and developmental disabilities (ID/DD). In particular there is growing recognition of autistic
spectrum disorders (ASD) and the implications for those affected (PHIS 2001, MRC 2001). Despite
government aims of developing appropriate services and supports (SE 2000), this study explored Scottish
mothers’ experiences and found continuing perceptions of unmet need.
Methods:
Mothers of 94 young people aged 13-22 in 4 groups:
1. typically developing controls (n=18)
2. with ID (n=37)
3. with ID, +ve ASD screening score and ASD diagnosis (n=19)
4. with ID, +ve ASD screening score but no ASD diagnosis (n=20)
All mothers completed standardised questionnaires on family and individual quality of life and child
behaviour. The mothers of young people with ID/DD also completed standardised questionnaires on stress
and service provision and a selected stratified sub-sample of 20 took part in semi-structured interviews
about stressful issues and perceived unmet need.
Results: Significant positive correlations were found between ASD screening score and maternal stress,
and significant negative correlations between family and individual quality of life and ASD screening score.
Significant differences were shown amongst the 4 groups in the study.
Themes emerging from interviews included: experiences of inadequate support, lack of social acceptance
and poor communication with and amongst service providers.
Discussion: Mothers of young people with ID may experience lower individual and family quality of life
than mothers of typically developing young people. Current developments aimed at service improvement
are consistent with mothers’ perception of need. These include: allocation of a key worker and improved
access to information and support. However, recent initiatives are not reaching all families who would
benefit. Whether this is because of poor implementation or misinterpretation of policies remains a subject
for further research.
References
MRC (Medical Research Council) (2001) Review of autism research: Epidemiology and causes, London,
Medical Research Council
PHIS (Public Health Institute of Scotland) (2001) Autistic spectrum disorders: Needs assessment report,
Glasgow, Public Health Institute of Scotland
SE (Scottish Executive) (2000) The same as you? A review of services for people with learning disabilities,
Edinburgh, The Scottish Executive
111
Pa p e r Se s s i o n 3
P A P E R
3
DOWN SYNDROME AND INTERVENTIONS
112
PAPER SESSION 3
Telomere Shortening in Chromosomes 21, 1, 2, and 16, and Reduced Signal Number in Tlymphocyte Metaphases from Older Individuals with Down Syndrome and Dementia
E.C. Jenkins1, M.T. Velinov1, L. Ye1, H. Gu1,Samantha A. Ni1, D. Pang1,2, D.A. Devenny1,
W.B. Zigman1, N. Schupf1,2, W.P. Silverman1,3.
1
NYS Inst Basic Res Dev Disab, Staten Island, NY;
2
Taub Inst Res on Alz Dis Aging Brain, Columbia Univ, NY;
3
Kennedy-Krieger Inst, Johns Hopkins Univ Sch Med, Baltimore, MD
Contact: [email protected]
Introduction: We have recently reported shorter telomeres (chromosome ends consisting of highly
conserved TTAGGG repeats) in T-lymphocytes from short-term whole blood cultures of women with Down
syndrome (DS) and dementia compared to age-matched women with DS but without dementia (NBA
2006;27:942-5). We have now accumulated a total of 13 people with trisomy 21 and dementia who have
exhibited shorter telomeres than their peers without dementia, including two male individuals. In addition
we have found four females with DS and without dementia but who exhibited what appeared similar to mild
cognitive impairment (MCI) in the general population, referred to here as MCI-DS. All had shorter
telomeres when compared to their non-demented peers. We also report our observations on telomere
shortening of individual chromosomes 21, 1, 2, and 16 as well as a reduced number of telomere signals in
metaphase chromosomes from people with DS and dementia.
Methods: Subjects were ascertained through the NYS Disability Service System and were classified as
demented by using parameters recommended by the Working Group for the Establishment of Criteria for
Diagnosis of Dementia in Individuals with Intellectual Disability (J Intellect Disabil Res 1997;41:152-64),
and methods are described in detail elsewhere (Am J Ment Retard 2004;109:111-25). Each subject was
age- and sex-matched such that one individual within each pair had dementia or MCI-DS and the other did
not. Twenty metaphases and 20 interphases per individual, as well as the individual chromosomes
mentioned above, were analyzed for telomere length differences by measuring changes in fluorescence
intensity using an FITC-labeled peptide nucleic (PNA) probe. Cells from age- and sex-matched individuals
with and without dementia or with and without MCI-DS were analyzed. In addition to the above
measurements, each metaphase chromosome arm was evaluated to determine how many chromosome
ends exhibited a fluorescent signal.
Results: Including our previous report, we have now observed shorter telomeres in metaphases and
interphases from 13 people with DS and dementia and 4 people with MCI-DS (p<.04-p<.000001). In
addition chromosome 21 was shorter in a total of 9 people studied with DS and dementia (4 others were
not studied for individual chromosomes) (p<.008-p<.000001) and in all four people with MCI-DS (p<.0002p<.000001). Shorter telomeres for chromosome 1 were also observed in 8 of 9 cases of people with DS
and dementia and for all four cases of MCI-DS. In four cases studied with DS and dementia all exhibited
shorter telomeres for chromosomes 2 and 16 and two other cases, with MCI-DS, also exhibited
chromosome two and 16 telomere shortening. Finally, we have observed an increased number of
chromosome ends with no fluorescent signal in nine of nine individuals analyzed, with DS and dementia
(p<.02-p<.000001) while two of four individuals with DS and MCI-DS also exhibited such an increase.
Discussion: We have now shown that shorter telomeres occur in people with DS and MCI-DS as well as
individuals with DS and dementia. In addition, we have shown that chromosome 21 may be used singly to
detect telomere shortening in individuals with DS and dementia or MCI-DS. Overall, there was no overlap
in the distribution of telomere length between demented and non-demented participants, t (19) = 6.5,
p<.0001 (and no overlap between participants with and without MCI-DS). The ability of these measures to
distinguish between cases with and without dementia was impressive. If further research confirms these
findings, they can provide the foundation for a diagnostic procedure having both high sensitivity and
specificity.
Source of Funding: In part by NYS OMRDD, Alzh. Assoc. grants IIRG-99-1598,IIRG-96-077; by NIH
grants PO1 HD35897, RO1 HD37425, RO1 AG014673, and RO1 AG14771.
113
PAPER SESSION 3
Down’s Syndrome, Ageing and Alzheimer’s Disease
*Holland, A.J., Ball, S. and Herbert, J.
University of Cambridge, UK
*[email protected]
Douglas House, 18b Trumpington Road, Cambridge, CB2 2AH, UK
Introduction: people with Down’s syndrome (DS) have a significantly reduced life-expectancy (McGrother
and Marshall, 1990) and age-related prevalence rates for Alzheimer’s disease (AD) that are similar to that
found in the general population but brought forward by 30 to 40 years, with prevalence rates reaching
between 50% and 60% in those 50 years and older (Lai and Williams, 1989; Holland et al, 1998).
Methods: as part of an on-going longitudinal study of older people with DS at risk for dementia physical
age-related markers and the level of the hormone, dehydroepiandrosterone ( DHEA), known to decrease
with age (Morales et al, 1994), were measured and compared to age-matched people without DS. The
hypotheses tested were that these measures in people with DS would be similar to that found in a
chronologically older population without DS, and that these age-related markers would be associated with
the risk of AD. Skin wrinkles, baldness and greying of hair, gait speed and hand grip strength were
assessed in 80 men and women with DS born before 1st July 1964 together with 36 controls in the same
age range, and a further 9 over the age of 70 years. DHEA blood levels were measured on 73 adults with
DS and 66 aged matched controls.
Results: all of the above physical markers and the DHEA levels were found to correlate significantly with
age in the expected direction in those with DS and controls. The most striking finding was that age-related
levels of DHEA were significantly lower in people with DS and may suggest an increase in ‘biological age’
of approximately 15 years, furthermore the level of DHEA significantly predicted dementia status – a level
of DHEA of one unit higher decreasing the odds of a diagnosis of dementia by 50%. In addition, within the
DS group, those with AD had significantly greater number of facial wrinkles compared to those without,
controlling for age.
Discussion: these findings give some support to the hypothesis that people with DS age prematurely and
that this may account for the reduced life-expectancy that is still observed. Its role in the causation of AD
requires further investigation but these findings, if confirmed, may have therapeutic implications.
Holland, A.J., Hon, J., Huppert, F.A., Stevens, F. and Watson, P. (1998) A population-based study of the
prevalence and presentation of dementia in adults with Down Syndrome. British Journal of Psychiatry,
172: 493-498
Lai, F. & Williams R.S. (1989) A prospective study of Alzheimer disease in Down syndrome. Arch Neurol,
46, 849-853
McGrother, C.W. & Marshall, B. (1990) Recent trends in incidence, morbidity and survival in Down’s
syndrome. J Ment Defic Res 34, 49-57
Morales, A.J., Nolan, J.J., Nelso, J.C. & Yen, S.C. (1994) Effects of replacement of
dehydroepiandrosterone in men and women of advancing age. J Clin Endo Metab., 78, 1360-1367
114
PAPER SESSION 3
Using Electropalatograpy (EPG) to Assess and Treat Speech Articulation Difficulties
in Children with Down’s Syndrome
Sara Wood1, Bill Hardcastle1, Joanne McCann1, Claire Timmins1, Jennifer Wishart2
1
Queen Margaret University College, Edinburgh, 2 University of Edinburgh
Contact: Dr Sara Wood
Speech Science Research Centre
Queen Margaret University College
Clerwood Terrace
Edinburgh EH12 8TS, Scotland, UK
[email protected]
Introduction: Speech development in Down’s syndrome (DS) is typically more impaired than would be expected
from levels of cognitive impairment. Articulation errors are common and often intractable. People with DS
often prefer visual to auditory methods of learning and may therefore find it easier to modify their speech
when given a visual-auditory rather than auditory-only model. EPG, a technique for recording and displaying
the timing and location of tongue-palate contact, provides just such feedback.
Methods: EPG is being used to assess and treat speech disorders in 30 children with DS
(10-18yrs), with data from 15 typically developing, MA-matched children as a control for task demands. All
participants are given a comprehensive battery of speech, language and cognitive assessments, including
EPG measures of articulatory accuracy and speech motor control. Participants with DS are then randomly
assigned to receive EPG therapy, articulation therapy or no therapy. Analyses are exploring the relations
between speech production and speech perception ability, cognitive level, chronological age and general
motor development, using pre- and post-therapy measures of intelligibility to evaluate intervention effects.
Results: Data from the first wave of testing will be presented (n =10). Children are presenting with mild to
severe speech disorders. EPG patterns are revealing many abnormalities undetected by an auditory analysis,
some of which are attributable to unusual, atypical processes rather than developmental delay. Examples of
such patterns include full anterior palatal contact for target /s/ accompanied by lateral release of air. Initial
data from the EPG-led therapy indicate that the children are able to correct abnormal patterns of contact
using the visual display offered by EPG.
Discussion: EPG may be an effective therapy for articulation disorders in children with DS, allowing more
successful communication with others and supporting a better quality of life.
Key References: Gibbon, F.E., McNeill, A.M., Wood, S.E. and Watson, J.M.M. (2003). Changes in
linguapalatal contact patterns during therapy for velar fronting in a 10-year-old with Down’s syndrome.
International Journal of Communication Disorders. 38, 47-64.
Roberts, J., Long, S., Malkin, C., Barnes, E., Skinner, M., Hennon, E. and Anderson, K. (2005). A
Comparison of Phonological Skills of Boys with Fragile X Syndrome and Down Syndrome. Journal of
Speech, Language, and Hearing Research. 48, 980-995.
115
PAPER SESSION 3
Inclusive Education and Social Isolation among Adolescents with Down Syndrome
Joanne Kersh, Penny Hauser-Cram, & Helena Pan
Boston College
Contact: Joanne Kersh, Boston College, Lynch School of Education,
Campion Hall, Chestnut Hill, MA 02467.
Email: [email protected]
Introduction: Preliminary results from the National Longitudinal Transition Study 2 suggest that high
school students with a range of disabilities have improved academic outcomes in inclusive educational
settings, when compared to earlier cohorts who were not in general education classrooms (www.nlts2.org/
). The benefits of inclusion at the secondary level are less clear, however, with regard to social outcomes.
Carter et al.(2005) found only moderate levels of social interaction among high school students with
intellectual disabilities and their general education peers, with greater interaction occurring in less
integrated settings. Their findings are troubling as low levels of social interaction may lead to feelings of
loneliness for students with disabilities. The focus of the current investigation is on whether individual
social skills or the school context (or both) relate to feelings of loneliness among adolescents with Down
syndrome (DS).
Methods: The current study is a secondary analysis of data from the Early Intervention Collaborative
Study (EICS), a longitudinal investigation of children with developmental disabilities and their families
(Hauser-Cram, Warfield, Shonkoff, & Krauss, 2001). The sample for this analysis consists of 26
adolescents (15-years old) with DS and their mothers. Teens were administered the Loneliness
Questionnaire (Williams & Asher, 1992) which measured their self-perceived feelings of loneliness and
social connectedness at school. The Socialization standard score of the Vineland Adaptive Behavior
Scales (mother report) was used as a measure of the child’s social skills. Mothers were asked additional
questions about their teen’s friendships and social activities and also asked to categorize their child’s
school day with regard to level of inclusion. For the purposes of this analysis, level of inclusion was
dichotomized into high inclusion (most instruction took place in the general education setting, with no more
than two special education classes) and low inclusion (the majority of the day spent in a special education
setting, with no more than two general education classes).
Results: Teens in the high and low inclusion groups did not differ from each other in terms of social skills
(F = .372, p = .548), adaptive behavior (F=.848, p = .366), or cognitive functioning (F=.263, p = .613). A
regression analysis was performed, using teen loneliness as the criterion variable. Neither teen social
skills nor level of inclusion were found to be significant main predictors; however, level of inclusion
moderated the relation between social skills and feelings of loneliness in teens with DS (t = 2.27; p =
.033). Specifically, teens with greater social skills reported fewer feelings of loneliness at school, but only
if they were in low inclusion environments. Teens with DS in high inclusion environments experienced
moderately high feelings of social isolation regardless of their level of social skills. This model explained
32.5% of the variance in loneliness among teens with DS overall. Additionally, 50% of the mothers of
teens in the high inclusion group reported that their child never socialized with other teens, compared with
only 13% of the mothers in the low inclusion group.
Discussion: These results suggest that while adolescents with DS in high inclusion settings may be
included in the general education curriculum, they do not feel included socially. Further, mothers’ reports
of their teens’ social experiences appear to validate the teens’ perspectives. We conclude that placement
in the general education environment is not, in and of itself, sufficient to promote social inclusion for
adolescents with DS. Suggestions for practice and policy are discussed.
Key References: http://www.nlts2.org/reports/changepop_report.html.
Carter, E.W., et al. (2005). AJMR,110, 366-377.
116
PAPER SESSION 3
Establishing the Building Blocks of Cognitive Behavioural Therapy with People with Mild
Learning Disabilities: Developing a Collaborative Relationship
Andrew Jahoda (University of Glasgow)*, Carol Pert (University of Glasgow), Peter Trower (University of
Birmingham), Biza Stenfert-Kroese (University of Birmingham), Dave Dagnan (University of Lancaster) &
Mhairi Selkirk (University of Glasgow)
*Department of Psychological Medicine, The Academic Centre, Gartnavel Royal Hospital, 1055 Great
Western Road, Glasgow G12 OXH, UK.
Contact: [email protected]
Background: There have been increasing efforts to adapt Cognitive Behavioural Therapy (CBT) for
people with mild intellectual disabilities. However, this is the first study to investigate whether a
collaborative relationship can be established between the therapists and clients, and the nature of the
clients’ understanding and experience of therapy.
Method: Sixteen clients with intellectual disabilities and depression, anxiety or significant problems with
anger were recruited from specialist community-based services offering CBT interventions for individuals
with dual diagnosis. A longitudinal design was adopted to investigate the nature of the therapeutic
relationship in the first phase of treatment. Ratings of adherence by an independent expert found the
therapists faithful to the CBT model. Data collection from each individual was extensive. This paper will
focus on therapy sessions 4 and 9, which were video and audio-taped in order to analyse the therapeutic
dialogue. The sessions were transcribed verbatim and the 12,544 communicative turns were then coded
using the innovative method of interactional analysis developed by Per Linell et al. (1988). This involved
coding each conversational partner’s turns in relation to the other, thereby making it possible to examine
the distribution of power in the dialogue (dominance).
Results: Analyses of relative dominance in the interaction between therapists and clients indicated a
reasonable balance of power, and that most clients played an active role in the dialogues. Yet it was found
that less able individuals produced more fragmented communication turns and most participants struggled
to grasp the CBT model. Therapists’ use of visual aids and active tasks, such as role-play, did help to
make complex components of therapy more meaningful.
Conclusions: In practical terms, the study findings promote understanding of the potential for people with
intellectual disabilities to engage successfully in CBT. The collaborative nature of the therapeutic
relationships contradicts the prevailing assumption that therapists should adopt a more didactic or
instructional approach to ensure engagement with individuals who have intellectual disabilities. The
limitations of the approach for this population are also highlighted.
References:
Linell, P., Gustavsson, L. & Juvonen, P. (1988) Interactional dominance in dyadic communication: A
presentation of initiative-response analysis. Linguistics, 26 (3), 415-442.
117
118
Po s t e r S e s s i o n 2
P O S T E R
2
THURSDAY, MARCH 8, 2007
6:30 P.M. - 8:30 P.M.
119
1.
POSTER SESSION 2
Language Development in Toddlers with Developmental Disabilities Following Intervention:
The Role of Child and Parent Communication Characteristics
R. Micheal Barker and Rose A. Sevcik
Georgia State University
One Park Place Suite 928, Atlanta, GA, 30303
[email protected]
It has been well documented that prelinguistic skills precede and may predict the subsequent emergence
of symbol use in children (e.g., Wetherby, Warren, & Reichle, 1998). This is true both for typically
developing children (Bates, Benigni, Bretherton, Camaioni, & Volterra, 1979; Bruner, 1983; Snow, 1977)
and children with developmental disabilities (Brady, Marquis, Fleming, & McLean, 2004; McCathren, Yoder,
& Warren, 1999; Mundy, Kasari, Sigman, & Ruskin, 1995; Yoder & Warren, 1999). Although much is
known about this relationship for these populations, relatively little is known about this process for very
young children with developmental disabilities who are just beginning to use language.
This poster will focus on two aspects of communication between young children with disabilities and their
parents. First, we will determine whether there is a connection between the gestures and pragmatic
functions children exhibit prior to a language intervention, and their performance on standardized
language measures post intervention. Second, we will determine whether parent communication
characteristics are predictive of child performance on standardized language measures above and beyond
the influence of child characteristics.
Participants were 59 children ranging from 24 to 36 months of age. All had significant developmental
disabilities, expressive vocabularies of less than 10 spoken words, and showed evidence of intentional
communication. Prior to intervention, each parent/child dyad participated in a 30-minute semi-structured
observation divided into three 10-minute segments. The observation session was structured such that it
had three distinct segments (i.e., play, book reading, and snack), designed to elicit a broad range of
communication. An observational coding scheme was utilized to characterize the communication
characteristics of children (i.e., mode, pragmatic function) and their parents (i.e., responsiveness) during
the baseline observation session. Participants were then randomly assigned to one of three 12-week
parent-led language interventions: Augmented Communication Input (ACI), Augmented Communication
Output (ACO), and Spoken Communication Intervention (SCI). The first intervention supported receptive
language in an augmented modality, the second supported expressive language in an augmented
modality, and the third supported spoken expressive language. Following the intervention, children were
administered an extensive assessment battery that included the MCDI and the SICD.
The results of three hierarchical regression analyses indicated that baseline child communication
characteristics accounted for a significant proportion of the variance in the MCDI receptive measure and
the SICD expressive and receptive measures. Children who used sophisticated gestures (defined as
symbolic and distal gestures) at baseline had significantly higher MCDI and SICD receptive scores than
those who did not. Children who used higher rates of sophisticated functions (defined as commenting,
requesting, attention getting, and answering) also scored higher on MCDI and SICD receptive measures.
Parent MLUm significantly predicted SICD receptive scores. Furthermore, children who used spoken
words (10 words or less) during the baseline assessment had significantly higher SICD expressive scores
than children who did not. Children who used sophisticated functions at higher rates had significantly
higher SICD expressive scores. A logistic regression revealed that children who used sophisticated
gestures at baseline were 14.7 times more likely to use greater than 10 words according to MCDI
expressive scores. Parent characteristics had no impact on these same expressive measures.
Implications for language development for children with significant disabilities will be discussed.
120
POSTER SESSION 2
2.
Repetitive Behaviors and Sensory Features in Young Children with Autism:
Examining Relationships and Specificity
Brian A. Boyd1, Grace T. Baranek, Linda R. Watson, & Elena Gay
Department of Allied Health Sciences, University of North Carolina at Chapel Hill
Introduction: It has been suggested in the field of autism that repetitive behaviors and sensory features
are inextricably linked or at the very least strongly related (Rogers & Ozonoff, 2005). Empirical research
has lagged behind theories on possible insults to shared neural systems or deficient neurocognitive
mechanisms that may underlie both classes of behavior. Even at the behavioral level little is known about
the relationship between repetitive behaviors and sensory features. Examining this relationship in children
with autism could provide insight into pathogenesis, shared or differential treatments for these classes of
behavior, or potentially have diagnostic implications for the third category of core autism features. The
purposes of this study are (a) to examine the relationship between sensory features and repetitive
behaviors in children with autism; (b) to identify differential associations for autism vs. a DD comparison
group that included children with Down syndrome; (c) to identify maturational variables (e.g., CA, MA, IQ)
that moderate any relationship; and (d) to examine their association(s) with other clinical features (e.g.,
symptom severity, adaptive skills).
Method: Data were collected from 66 children with autism (n=48) and DD (n=18), ages 2-7, using
empirically-valid parent and observational measures. Parent-report sensory measures included the
Sensory Experiences Questionnaire (SEQ) & Sensory Profile (SP). Observational sensory measures
included the Sensory Processing Assessment (SPA) & Tactile Defensiveness & Discrimination TestRevised (TDDT-R). Derived factor solutions from the Repetitive Behavior Scales-Revised (RBS-R) (parent
report measure) were used to analyze the repetitive behavior data (Lam & Aman, 2006).
Results: We have run preliminary correlation analyses with the autism and DD groups combined.
Analyses indicate the stereotypy factor on the RBS-R is most strongly correlated with the sensory parent
report (SEQ, SPA) and observational (SPA only) measures. The correlation between children’s total mean
SEQ score and stereotypy is 0.73 (p<.0001), and only the stereotypy subscale is correlated with the SPA.
The 4 remaining RBS-R subscales yielded low to moderate correlations. After controlling for children’s IQ
scores based on the Leiter, significant correlations maintained between parent report sensory measures
and the RBS-R. Prior to the poster session we will run additional analyses to parse out differences for the
autism vs. DD groups with additional subjects added to each group. Further, items from the 4 sensory
measures will be conceptually grouped into hyporesponsive, hyperresponsive, and sensory seeking
constructs to examine their relationship to the 5-factor solution derived for the RBS-R. We have found
stronger correlations between CARS’ total score (measure of autism severity) and mean scores on the
SEQ hyporesponsiveness (0.54, p=.00) vs. hyperresponsiveness (0.25, p=.04) subscales for our current
sample, with stereotypy again being the repetitive behavior factor most correlated with the CARS (0.42,
p=.00).
Discussion: The preliminary results indicate that sensory features in children with autism and DD are
related to repetitive behaviors. In particular, the presence of stereotypy appears to be most strongly
associated with sensory features. Autism severity appears to be more strongly related to
underresponsiveness vs. overresponsiveness to sensory stimuli as well as presence of stereotypy.
Supported by: NIH HD42168
1
Contact:
Brian Boyd, Ph.D.; Email: [email protected]; Mailing Address: UNC-Chapel Hill, Department of Allied Health
Sciences, CB# 7122, Chapel Hill, NC 27599-7122; Phone: 919.843.2628
121
3.
POSTER SESSION 2
Predicting the Depression and Well-being of Mothers Rearing
Children with Developmental Disabilities
April M. Corrice, Laraine M. Glidden, and Brian M. Jobe
St. Mary’s College of Maryland, Department of Psychology
St. Mary’s City, MD 20686
Contact: [email protected]
Because birth mothers had significantly higher depression scores at the time of their children’s diagnoses
of developmental disabilities (DD) than adoptive mothers at the time of their children’s placement, Glidden
and Schoolcraft (2003) were interested in similarities and differences in later functioning between these
adoptive and birth mothers when their children were, on average, 11 years old. Glidden and Schoolcraft
reported that concurrent scores for the personality variable of Neuroticism powerfully predicted depression
for all mothers. For birth mothers only, earlier depression scores predicted later depression and subjective
well-being related to the child was predicted by concurrent scores for the personality variable of
Agreeableness and earlier levels of family accord. The current study extends the findings of Glidden and
Schoolcraft and introduces new variables with the potential to predict depression and subjective well-being
related to the child. Adoptive or birth status was also examined as a potential predictor of maternal
functioning.
Four times of measurement, each separated by 5-6 years, were included in this longitudinal study. The
Family Strengths Scale (FSS), Questionnaire on Resources and Stress (QRS), NEO Five-Factor Inventory
(NEO-FFI), and Adaptive Behavior Scale (ABS) served as predictors of the Beck Depression Inventory
(BDI) and Subjective Well-being, Child-related (SWB-CR). Mothers completed each measure, except for
the ABS, which was completed by the children’s teachers. The final sample included 123 mothers (69
birth and 54 adoptive) who were part of an original sample consisting of 249 families with at least one child
with DD.
BDI and SWB-CR mean scores were low for both adoptive and birth mothers, demonstrating positive
functioning. Specifically, mean depression scores were in the non-depressed range and mothers were, on
average, “pleased” to “mostly satisfied” with how things were going with their children. Significant
predictors of BDI and SWB-CR scores were determined via multiple regressions. Time 2 BDI, time 3 BDI,
and FSS Family Accord were uniquely significant predictors of time 4 BDI. These results indicate that
depression is related to itself over time and that the less conflict families experienced and the better they
were able to resolve disputes, the less future maternal depression. Earlier SWB-CR, NEO-FFI Openness,
and QRS Personal Burden predicted later SWB-CR, demonstrating that maternal well-being regarding the
child is related to itself over time. Also, mothers who score high on Openness may be more understanding
and accepting of their children’s differences, which could contribute to more positive subjective well-being
related to the child. Lastly, the less burdened mothers are, the greater their well-being related to the child.
Neither NEO-FFI Neuroticism nor adoptive/birth status were significant predictors of BDI or SWB-CR
scores.
In conclusion, these results demonstrate that, overall, both adoptive and birth mothers manifest good
adjustment, consistent with the findings of Glidden and Schoolcraft. Also, the mothers’ functioning is
closely intertwined with individual and family variables. Thus, intervention at an individual or family level
may be effective for those mothers who do experience depression or low levels of subjective well-being
related to the child.
Reference
Glidden, L. M., & Schoolcraft, S. A. (2003). Depression: Its trajectory and correlates in mothers rearing
children with intellectual disability. Journal of Intellectual Disability Research, 47, 250-263.
122
4.
POSTER SESSION 2
Positive States as Mediators of Stress in Parents of Children with Different Types of
Intellectual Disabilities
Elizabeth Roof, Elizabeth Pantino, Rebecca Kossler, Ray Johnson, & Elisabeth M. Dykens
Vanderbilt Kennedy Center, 230 Appleton Place, Nashville, TN 37203
[email protected]
Introduction: Although parenting children with intellectual disabilities is consistently associated with
heightened stress, several child and parent factors appear to mediate stressful outcomes. Maternal stress
is associated with heightened child maladaptive behaviors, as well as the child’s etiology. For example,
stress is generally higher in mothers of children with Prader-Willi syndrome and with autism as compared
to families of children with Down syndrome. Mothers who adopt problem-solving coping styles fare better
than those who do not. Studies have not yet examined the effects of positive parental perceptions on
parental stress, nor have they routinely used cortisol levels as a biomarker of stress. This study examined
relations among parental stress, anxiety, coping, health status, and cortisol levels. Positive perceptions of
raising a child with disabilities were cast as a protective factor that would predict lower stress and cortisol
levels, as well as improved health.
Methods: Parents of 30 children with Prader-Willi sydrome, 30 with Williams syndrome, and 40 with
autism spectrum disorders completed a health survey; Parenting Stress Index; Beck Anxiety and
Depression Scales; Parental Ways of Coping; Positive Parenting and Positive Perceptions; and child
maladaptive behavior. Cortisol levels were extracted from 6 saliva samples collected at set intervals over
a 24-hour period. Parents ranged in age from 30 to 62 years, with mean of 43 years in each group, the
majority of respondents were mothers; 20% in each group were fathers.
Findings: Compared to parents of children with Williams syndrome, significantly higher levels of stress
were found in parents of offspring with Prader-Willi syndrome and with autism spectrum disorder (p < .01).
Significant, inverse relations emerged between positive perceptions of raising a child with disabilities and
levels of parental stress, depression, and anxiety. Cortisol samples are currently being analyzed in the
Williams syndrome and autism groups. Within Prader-Willi syndrome, fathers had significantly higher
cortisol levels than mothers. Maternal cortisol levels were positively associated with seeking social
supports, specific health concerns, and number of offspring (r’s = .64 to .67, p’s < .01).
Discussion: Etiology and positive perceptions both affect levels of parental stress, and cortisol levels
hold promise as a biomarker of stress responses for parents of children with intellectual disabilities.
123
5.
POSTER SESSION 2
Insecure and Disorganized Attachment among At-Risk Children:
The Influence of Subthreshold Neglect
Jaelyn R. Farris, Jody S. Nicholson, and John G. Borkowski
University of Notre Dame
The security and organization of infant-mother attachment bonds are at risk when caregivers fail to respond to their children in a responsive and sensitive manner. Insecure and disorganized attachment may
facilitate the early onset of developmental delays, especially youth psychopathology. Certain factors, such
as adolescent parenting, may increase the likelihood of unresponsive or neglectful caregiving and, in turn,
jeopardize the formation of a secure and organized attachment bond. In addition, children of adolescent
mothers are at risk for developmental delays. Although it has been established that adolescent parenting
and child neglect are associated with insecure and/or disorganized attachment, less is known about: (1)
the effects of “subthreshold neglect,” in which parents provide unresponsive and/or inattentive care that is
severe enough to put children at risk for maladjustment but less severe than the legal criteria used to
mandate intervention, (2) the unique effects of subthreshold neglect in various domains (e.g., physical,
emotional, and educational), and (3) variations in these relationships between adolescent versus adult
mother-child dyads. The present study focuses on the impact of subthreshold physical, emotional, and
educational neglect on the security and organization of infant-mother attachment at one year of age
among adolescent and adult mother-child dyads.
Participants are 263 primiparous mothers and their children drawn from a larger longitudinal project.
Approximately half (53.2%) of the sample is comprised of adolescent mothers. The sample is racially
diverse, with approximately 65% African-American, 19% European-American, and 14% Latina mothers;
these rates are nearly equivalent between teen and adult groups. Measures of subthreshold physical,
emotional, and educational neglect were assessed when infants were 4 and 8 months of age through
home-based interviews and observations using the Landry Observation of Parent-Child Interactions, the
Infant/Toddler Home Observation for the Measurement of the Environment, the Supplement to the Home
scale for Impoverished Families, and interviewer ratings of neglectful behaviors. Attachment between
mothers and children was assessed in the lab at 12 months; videotapes of the Strange Situation are
currently being coded by a team of four raters who were trained and certified through the Institute of Child
Development at the University of Minnesota.
The analyses for this study, which are underway pending final coding of attachment data, consist of three
phases: (1) the development of scales that represent subthreshold physical, emotional, and educational
neglect, (2) a description of the prevalence of A, B, C, and D attachment classifications, and (3) the prediction of attachment classifications from subthreshold physical, emotional, and educational neglect. It is
expected that multiple indicators from the home-based measures will represent the latent constructs of
subthreshold physical, emotional, and educational neglect; that the rates of insecure and disorganized
attachment will be elevated among children of adolescent mothers; that children exposed to more severe
levels of subthreshold neglect will be more likely to have insecure and disorganized attachment patterns;
and that subthreshold emotional neglect will be a stronger predictor of insecure and disorganized attachment than subthreshold physical or educational neglect. Findings from this study will have the potential to
inform future prevention and intervention work, enabling programs to target specific domains of unresponsive parenting, rather than an overall neglect construct. Resultant programs may facilitate secure and
organized infant-mother attachment relationships and prevent the onset of developmental delays, such as
youth psychopathology, that may be associated with insecure and/or disorganized infant-mother attachment patterns.
124
6.
POSTER SESSION 2
The Role of Caregiver Praise as a Mechanism for Children’s Sustained Engagement
Kim Har & Connie Kasari
University of California, Los Angeles, Graduate School of Education
Contact: Kim Har, UCLA Graduate School of Education,
Moore Hall, Box 951251, Los Angeles, CA 90095-1521
Contact: [email protected]
Previous literature reports that children with Down syndrome tend to exhibit lower levels of sustained
engagement during interactive tasks than typically-developing peers (e.g. Cielinski, Seifer & Contreras,
1995). Meanwhile, studies have found that the caregivers of children with Down syndrome are more
supportive and provide more praise during interactive tasks than caregivers of typically-developing
children (Roach, Barratt, Miller, & Leavitt, 1998; Hughes & Kasari, 2000). The current study attempts to
explore these two themes by examining the nature of caregiver-praise and the sequential way in which it
may be exhibited; specifically, the study asks: do support and praise from caregivers serve as a
mechanism to help maintain the interest and continued engagement of children with Down syndrome
during interactive tasks? Twenty children with Down syndrome and their caregivers participated in the
current study, along with 20 typically-developing children and their caregivers. The two groups were
matched based on their levels of mental age. The children with Down syndrome (mental age 40.25)
ranged from four to eight years of age, while the typically-developing children (mental age 39.65) ranged
from 30 months to four years of age. Child-caregiver dyads were asked to engage with toys that had clear
criteria for completion (e.g., puzzles). These videotaped interactions were then coded for child response
and engagement, as well as caregiver praise. Inter-rater reliability was found to be good (G=0.73) to high
(G=0.91). Group differences in child and caregiver behavior between the Down syndrome and typical
group were examined during the process of completing the task and at task completion. Caregivers of
children with Down syndrome provided more frequent praise during task completion than did caregivers of
typical children. Children with Down syndrome were also found to exhibit greater social orientation to their
caregivers than the typically-developing children, F(2,76) = 5.40, p < .05; however, during the interaction,
they were also more frequently disengaged from the task itself. Secondary analyses are now being
conducted to examine associations between caregiver and child behaviors in this task and in a story book
reading task. Initial results indicate that, for group with Down syndrome, caregiver praise is directly
associated to continued engagement during both interactive tasks. Additional analyses involving
sequential coding of caregiver praise and children’s responses will reveal further insight concerning the
nature of the relationship between caregiver praise and sustained engagement of children with Down
syndrome. These results, suggesting that caregiver praise might play a facilitating role in maintaining the
interest and active engagement of children with Down syndrome during learning tasks, may have potential
implications for special educators and families of children with Down syndrome.
References
Hughes, M. & Kasari, C. (2000). Caregiver-child interaction and the expression of pride in children with
Down syndrome. Education and Training in Mental Retardation and Developmental Disabilities, 35, 67-77.
Roach, M.A., Barratt, M.A., Miller, J.F., & Leavitt, L.A. (1998). The structure of mother-child play: Young
children with Down syndrome and typically developing children. Developmental Psychology, 34, 7787.
Cielinski, K.L. & Vaughn, B.E. (1995). Relations among sustained engagement during play, quality of play,
and mother-child interaction in samples of children with Down syndrome and normally developing
toddlers. Infant Behavior and Development, 18, 163-176.
125
7.
POSTER SESSION 2
Stressful Social Interactions, Attributions, and Coping among Depressed Adults with Mild
Intellectual Disability-Poster Presentation
Sigan L. Hartley* and William E. MacLean, Jr.
*Corresponding Author: University of Wyoming, Department of Psychology,
Dept. 3415, 1000 E. University Ave, Laramie, WY 82071
Email: [email protected]
Introduction: Depression is one of the most frequently encountered mental health problems among
people with intellectual disability (ID). Little is known about the behavioral, cognitive, and interpersonal
variables related to the onset, presentation, and persistence of depression in an ID population. In the
general population, depressed persons frequently experience interpersonal stress, endorse negative
attributions for stressful events (i.e., internal, stable, and global causes), and utilize maladaptive coping to
deal with these events. These processes are often the focus of therapies and are theorized to contribute to
the onset and persistence of depression over time. Whether interpersonal stress, negative attributions,
and maladaptive coping are prominent features of depression among adults with ID is unknown.
Methods: The frequency and stress impact of stressful social interactions, causal attributions for these
interactions, and coping strategies of 31 depressed and non-depressed adults with mild ID matched on
various subject characteristics were examined. Participants were recruited through 10 disability services
providers in the Rocky Mountain region. The Diagnostic Criteria for Psychiatric Disorders for Use with
Adults with Learning Disabilities/Mental Retardation (Royal College of Psychiatrists, 2001) and Birleson
Depressive Short Form Self-Rating Scale (Birleson, 1981) were used to confirm diagnoses of depression.
Self-report measures were used to assess the frequency and stress impact of stressful social interactions.
Open-ended questions were used to elicit attributions for stressful social interactions and coping efforts
and responses were coded by two undergraduate students who were naïve to the study hypotheses.
Results: Depressed adults with mild ID reported a higher frequency and stress impact of stressful social
interactions than non-depressed adults. Depressed adults with mild ID attributed stressful social
interactions to more internal, stable, and global causes than non-depressed adults. Depressed adults with
mild ID also reported fewer active coping strategies, particularly Support Seeking coping, and more
avoidant coping, particularly Cognitive Avoidance coping, than the non-depressed adults.
Discussion: Findings have implications for understanding and treating depression in an ID population.
Depression in adults with mild ID may be partially maintained over time by heightened rates of stressful
social interactions, maladaptive coping efforts, and tendencies to attribute conflict with others to internal,
stable, and global causes. Psychotherapies that alter these maladaptive cognitive tendencies and
interpersonal skill deficits, and encourage active coping may be appropriate interventions for depression.
126
8.
POSTER SESSION 2
Family Cohesion and Its Influence on Adaptive Behavior Development in
Children with a Disability
Lara S. Head
Waisman Center, University of Wisconsin-Madison
1500 Highland Ave., Suite 533, Madison, WI 53705
Contact: [email protected]
Alan M. Gross
The University of Mississippi
Introduction:
Family cohesion represents a potentially critical missing explanatory construct in much of the research
examining how families interact and function in response to raising a child with a disability. Although such
families are at risk for the development of dysfunctional family interaction patterns, little is known about the
impact of family functioning on the adaptive behavior development of children with developmental
disabilities. This study examined the relationship between family cohesion and adaptive behavior
development in children with a disability, hypothesizing that children in families with high levels of cohesion
would demonstrate better developmental outcomes in adaptive behavior as compared to children in
families reporting lower levels of cohesion.
Method:
Forty-two parents of children diagnosed with a developmental disability were compared with 43 parents
and their typically developing children. Children ranged in age from 7 and 11 and were matched on age
and gender. Control child participants were assessed using the Peabody Picture Vocabulary Test –III for
estimated average cognitive functioning. Special education records were used for experimental group
inclusion based on diagnosis of mental retardation and/or other identified developmental disability.
Parents of both groups of children completed self-report measures assessing their perceptions of family
interaction and functioning. Parents of children with a disability also completed the Vineland Adaptive
Behavior Scales – Interview Edition to assess global and specific areas of adaptive behavior functioning in
their children.
Results:
Findings demonstrated that families with a child with a disability did not significantly differ in cohesion as
compared to families with a typically developing child. A positive relationship was observed, however,
between cohesion demonstrated within the family as a unit and levels of cohesion within the marital or
partner relationship. Finally, controlling for IQ, parental stress, was the greatest predictor of global
adaptive development in the children (R = .915, R² = .837, Adjusted R² = .822, ΔR2 = .072, F (1, 32) =
14.253, p = .001).
Discussion:
Results suggest that families with a child with a disability may be functioning as effectively as families with
typically developing children in terms of cohesiveness. Furthermore, the positive interactions
characteristic of cohesive families may be protecting families from the negative influences of stress
associated with caregiving and parenting a child with a disability. Rather than such challenges having a
negative impact, they may serve as a catalyst for the development of a level of cohesion that meets the
needs of the family.
Key References
Hauser-Cram, P., Warfield, M. E., Shonkoff, J. P., & Krauss, M. W. (2001). Children with disabilities: A
longitudinal study of child development and parent well-being. Monographs of the Society for Research in
Child Development, 66, 1-131.
127
9.
POSTER SESSION 2
Father Presence as a Predictor of Maternal Stress and Quality of Life in
Families with Children with Fragile X Syndrome
Anna James, Jane E. Roberts, and Don Bailey
University of North Carolina
517 South Greensboro Street, Chapel Hill NC 27599-8040
Contact: [email protected]
Introduction: Evidence shows that fathers provide a unique and important contribution not only to their
children’s development but also to the family’s well being. Despite an upsurge in interest and recognition
of the significance of the father’s role, research investigating the importance of father presence in families
with children with disabilities has been scarce. Studies indicate that mothers who take on the bulk of caring
for a child with a disability experience more stress and often rely on others for support. In particular,
evidence suggests that mothers of children with disabilities report needing practical assistance more than
emotional support from their partners. Taken together, this suggests that a father’s presence may help
relieve some of the burden of care; thus, improving the mother’s well being. The purpose of this study is to
explore the extent to which fathers’ presence is related to maternal stress and quality of life in families with
children with fragile X syndrome (FXS).
Method: The data presented in this poster were derived from an ongoing longitudinal study of adaptation
in families with a child with FXS. One-hundred-and-four mothers are included in this sample. Eighty-two
mothers reported a father present in the home, and 22 of the mothers reported that there was no father
present. All of the target children had the full mutation of FXS. The Parenting Stress Index-Short Form
(PSI) and the Quality of Life Inventory (QOLI) were completed by each mother to collect information on
perceived stress resulting from the parental role and life satisfaction. Family demographic information was
utilized to group participants into two groups: 1) mothers who reported a father present in the home, and
2) mothers who reported a father not present in the home.
Results: Covarying for household income, analyses of variance (ANOVA) revealed elevated stress in
mothers without the father present in the home for the Parental Distress (F(3, 100) = 15.07, p = <.001)
and Defensive Responding (F(3, 99) = 13.29, p = <.001) subscales as well as for the Total Parenting
Stress composite (F(3, 100) = 9.15, p = <.01). Lower quality of life scores were found in mothers without
the father present in the home (F(3, 98) = 14.79, p = <.001). No effects were found for household income
or for household income-by-father presence interaction.
Discussion: Preliminary analyses of our data indicate that fathers’ presence is important for maternal
perceptions of parenting stress and quality of life in families with children with FXS. Such findings are
important to the development of policy to optimize the well being of single mothers of children with
disabilities. Some research suggests that the effects of fathers’ presence could be moderated by child
problem behaviors and that mother’s satisfaction with the father’s role is a more important predictor for
their well being than the father’s actual presence. Therefore, additional investigation is needed to delineate
the specific role father presence plays in decreasing maternal parenting stress and improving quality of
life.
128
10.
POSTER SESSION 2
Auditory Brainstem Evoked Responses in Newborns with Down Syndrome
P. Kittler1, 2, B.Z. Karmel1, 2, H.T.T. Phan1, 2, J.M. Gardner1, 2, A. Gordon1, 2, & A. Harin2
1
NYS Institute for Basic Research in DD & 2St. Vincent’s Catholic Medical Center, Staten Island, NY email:[email protected]
Auditory brainstem evoked responses (ABRs) have been used to help understand and localize the hearing
impairment and poor auditory processing that is widespread among the population with Down syndrome
(DS). A common finding in this group has been shortened peak component latencies (CLs), particularly of
Wave V1, 2. Previous studies generally have included broad age ranges, with few participants under 1 year
of age. The present study examined ABRs in neonates with DS and compared them to ABRs in infants
without DS.
Participants were an opportunistic sample of all newborns diagnosed with DS and administered ABRs
(n=27), during the last 9 years of a longitudinal study of attention and arousal in high risk infants3. Twenty
of 27 newborns with DS had normal ABRs, and were matched by sex, postconceptional age (PCA) at test,
and birth weight to 20 newborns with normal ABRs and without DS. This group of 40 participants was 65%
female, had a mean birth weight of 2643 g, and a mean PCA at test of 37.8 weeks. Despite matching on
birth weight and PCA, head circumference was smaller in the group with DS (F (1,38)= 4.12, p<.05).
Procedure Ipsilateral left ear vertex-to-mastoid recordings were
made using 100 µsec rectangular rarefaction click stimuli
presented at 12.1 Hz through a miniature earphone at 80dB re
adult nHL. A composite ABR consisting of the average of 3 x
1024 artifact-free sweeps was used to identify CLs. Stimulus
delivery and response averaging were done with Intelligent
Hearing System Smart EP hardware and software modules.
Testing occurred within a short time after birth (M=5.1 days) in
the neonatal intensive care unit or newborn nursery. Previously
established laboratory normative values, specific to PCA, were
used to categorize ABRs as normal or abnormal4.
Wave V Peak Latencies
Latency in msec
7.5
7
6.5
6
5.5
31
33
35
37
39
41
Results Comparisons between the DS and control groups
indicated no difference in Wave I CLs, but both Wave III (F
=12.32, p<.001) and Wave V (F(1,38)= 13.55, p<.001) CLs
DS Control
(1,38)
were significantly shorter for neonates with DS.
Discussion These results extend findings of shortened ABR Wave III and V CLs in DS to the earliest
newborn period. Shortened latencies in DS neonates suggest that the neuroanatomical basis for this
difference emerges very early in development. The patterns observed here are similar to those recently
reported among newborns with intrauterine growth retardation5. Whether the etiology of this ABR pattern
for the neonates with DS and those with intrauterine growth retardation are similar remains to be
investigated.
PCA at test (w ks)
References
1
Seidl, R. et al. (1997.) Auditory evoked potentials in young patients with Down syndrome. Event-related potentials (P3) and histaminergic system. Cognitive Brain
Research, 5, 301-309.
2
Krecicki, T. et al. (2005). Brain auditory evoked potentials in children with Down syndrome. International Journal of Pediatric Otorhinolaryngology, 69, 615-620.
3
Gardner, J.M. et al. (2006). Arousal, attention, and neurobehavioral assessment in the neonatal period: implications for intervention and policy. Journal of Policy
and Practice in Intellectual Disabilities, 3, 22-32.
4
Karmel, B.Z. et al. (1988). Brain-stem auditory evoked responses as indicators of early brain insult. Electroencephalography and Clinical Neurophysiology, 71,
429-442.
5
Phan, H.T.T. et al. (2006, Oct). Auditory brainstem evoked responses in newborn twins. 2006 Meeting of the International Society for Developmental
Psychobiology, Atlanta, GA.
This work is supported by NICHD grant P01-HD47281, R01- HD21784, and NYS OMRDD
129
11.
POSTER SESSION 2
Gender and Delay of Gratification in High Risk Children
Shira Kolnik ([email protected].edu), Ann-Marie Faria ([email protected]),
Marygrace Yale Kaiser ([email protected])
University of Miami, 5665 Ponce de Leon Blvd, Coral Gables, FL 33124
Previous studies have examined the ability to delay rewards for a period of time in order to attain a better
reward (e.g., Mischel, 1966; Mischel & Ebbesen, 1970). The ability to delay gratification is associated with
numerous outcomes including lack of resilience, addictive disorders, and antisocial behavior (Mischel,
Shoda & Rodriguez, 1989). A meta-analysis of gender differences in delay of gratification found that
females are better at delaying rewards than males, especially when continuous measures were used
(Silverman, 2003). The meta-analysis was restricted to studies of normal samples.
There is a lack of published articles studying delay of gratification in at risk or special needs populations.
The current study samples from a population of children previously enrolled in a birth-to-three intervention
for prenatal exposure to cocaine. The sample of 19 children is primarily African American and low socioeconomic status, placing them at risk for behavior problems (Arnold & Doctoroff, 2003). Participants range
from 6 to 8 years of age, with 9 females.
The delay of gratification paradigm (Mischel, 1966) was used to measure ability to delay rewards by
placing two choices in front of the child, one plate with a small pile of food and one plate with a larger pile.
The child is told that he can have the small pile of food whenever he wants, but that if he waits seven
minutes without eating the food he will receive the larger pile. The child is left to wait in the room for up to
seven minutes. Eating the food or ringing a bell will terminate the session, leaving the child with the
smaller pile of food.
Two analyses of the continuous measure of seconds spent waiting reveal a gender effect with males
waiting longer than females. This is opposite to the gender effect that has previously been reported in the
literature with females waiting longer than males in typical populations. An analysis of variance revealed
that boys waited significantly longer than girls (Mmales=390, Mfemales=212, F(1,17)= 10.57, p<.01). A
post-hoc analysis of effect size and power found a large effect size (Cohen’s d=1.49) and adequate power
(power=.8). A multivariate linear regression revealed gender as a significant predictor of time spent
waiting (β=248.16, p<.01). A Chi-square statistic of dichotomous outcomes, whether the child waited the
entire time or terminated the session, showed significantly more males waiting the entire session than
females (x2(1)=6.357, p<.01).
The data raises doubt about the generalizability of the gender effect to special needs populations. Contrary to reported gender effects in typically developing populations, the gender effect in this high risk
population of children shows a strong gender effect of males waiting longer than females. This effect held
true for both dichotomous and continuous measures of delay of gratification. Subsequent studies should
attempt to replicate the findings with larger samples of at risk and special needs populations.
Arnold, D.H. & Doctoroff, G.L. (2003). The early education of socioeconomic disadvantaged children. Annual Review
of Psychology, 54, 517-545.
Mischel, W. (1966). Theories and research on antecedents of self-imposed delay of reward. In B. Maher (Ed.),
Progress in experimental personality research. New York: Academic Press.
Mischel, W. & Ebbeson, E.B. (1970). Attention in delay of gratification. Journal of Personality and Social Psychology,
16(2), 329-337.
Mischel, W., Shoda, Y. & Rodriguez, M. L. (1989). Delay of gratification in children. Science, 24(4907), (933-938).
Silverman, I. W. (2003). Gender differences in delay of gratification: A meta-analysis. Sex Roles, 49, (451-163).
130
12.
POSTER SESSION 2
Changes in Joint Attention Following Two Years of Early Intensive Behavioural
Intervention in Preschool Aged Children with Autism
Hanna Kovshoff1, Richard Hastings2 & Bob Remington1
1
School of Psychology, University of Southampton, UK
2
School of Psychology, University of Wales, Bangor, UK
Contact: [email protected]
Introduction: Children with autism are often reported to be significantly delayed or deviant in their
development of precursors to social communication including eye contact, sharing attention, and pointing
out items of interest. The present study monitored the longitudinal development of joint attention in a
group of preschool aged children with autism who received Early Intensive Behavioural Intervention (EIBI)
(n = 21) and a Comparison group (n = 16) who received a range of local educational authority provision in
the South of England across two years. The aim was to identify whether EIBI was able indirectly to target
and improve the initiation or response to joint attention in the group of children who received it.
Method: All of the children were assessed using the Early Social Communication Scales (ESCS) (Mundy,
Hogan, & Doehring, 1996) at three time points (baseline, 12 month follow-up, and 24 month follow up).
The ESCS is a videotaped structured observation measure that provides an index of individual differences
in nonverbal communication skills.
Results: After two years of EIBI, children in the Intervention group made significant gains in their ability to
respond to joint attention (RJA) relative to the Comparison group. Initiating joint attention (IJA) increased
in frequency for both groups over time but significant group differences were not found.
Discussion: While RJA may increase as a concomitant effect of engaging in early intervention, the ability
to initiate joint attention may need to be targeted directly to promote positive change relative to a
Comparison group. Further research is needed to evaluate whether direct targeting of IJA may
compensate for a difficulty in generalizing from structured teaching situations. Measurement and
assessment of joint attention in children with autism will also be discussed.
131
13.
POSTER SESSION 2
Maternal Predictors of Adaptive Behavior of Boys with Fragile X Syndrome
Heather Magruder, B.A.
FPG Child Development Institute, University of North Carolina at Chapel Hill
517 S. Greensboro St; Carrboro, NC 27510-2341
Contact: [email protected]
Introduction: Adaptive behavior refers to both practical (self-care, work, and play) and social (interaction
and communication) constructs that describe how well an individual can function independently (American
Association on Mental Retardation, 1992). Research suggests that chronological age (CA), gender,
severity of intellectual disability, number of autistic behaviors, and screenings of current maternal
depressive symptoms are significant predictors of adaptive behavior of boys with FXS. However, few
researchers have examined the effect that lifetime occurrence of major depressive disorder (MDD) has on
the adaptive behavior of boys with FXS. The purpose of this study was to address this gap in the literature
by examining the effect of lifetime occurrence of MDD, maternal parenting stress, and martial intimacy on
adaptive behavior skills in boys with FXS while controlling for CA and autistic behavior.
Method: This study included 47 families with a boy with the full mutation of FXS and mothers who
reported a spouse in the home. Due to the focus of this study on MDD, mothers with MDD (with or without
co-morbid anxiety disorders) were included (n = 24) as were mothers with neither mood nor anxiety
disorders (n = 23). The age of the boys ranged from 9 months to 8.5 years (M = 36.57 months). Adaptive
behavior was measured using the Vineland Adaptive Behavior Scales, Interview Edition; autistic behavior
was assessed using the Childhood Autism Rating Scale (CARS); MDD was diagnosed using the
Structured Clinical Interview for DSM-IV Axis I Disorders- Research Version; maternal parenting stress
was measured using the Parenting Stress Index- Short Form (PSI); maternal marital intimacy and support
was measured by the Emotional Intimacy subscale of the Personal Assessment of Intimate Relationships
Inventory (PAIR).
Results: A univariate general linear model was run to examine maternal predictors of adaptive behavior.
The Vineland Composite Standard Score was the dependent variable and a history or MDD, PSI, and
PAIR scores were the predictor variables. Chronological age and autistic behavior were covariates.
Results suggest a significant interaction between a history of MDD, PSI, and PAIR scores in predicting
Vineland Composite scores, F(1,38) = 5.60, p = .023. The strength of the interaction was fairly modest and
accounted for 13.2% of the variance of the Vineland Composite Scores.
Discussion: The results of this study suggest that a history of maternal MDD, current maternal parenting
stress, and current marital intimacy and support all contribute to adaptive behavior skills in young boys
with FXS. There are several limitations of this study. First, the directionality of the observed effect cannot
be determined because some of the major depressive episodes occurred before the birth of the child,
while others occurred after. Second, some of the women with MDD also had an anxiety disorder that could
have had an effect on their children. However, the sample sizes are too small to test this relationship
directly. Finally, we did not consider the severity, duration, or number of depressive episodes, all of which
could affect adaptive behavior differently.
Reference:
American Association on Mental Retardation (1992). Mental Retardation: Definition, Classification, and Systems of
Support, 9th ed. Washington, DC: American Association on Mental Retardation.
Kessler, R. C., Berglund, P., Demler, O., Jin, R., Merikangas, K. R., & Walters, E.E.
(2005). Lifetime prevalence and age-of-onset distributions of DSM-IV disorders in the National Comorbidity
Survey Replication. Archives of General Psychiatry, 62, 593-602.
132
14.
POSTER SESSION 2
Autism in Early Childhood: Identification and Treatment
Nicole J. Neudorfder, Aila Dommestrup, & Laura Lee McIntyre
Syracuse University
Contact: Laura Lee McIntyre
Department of Psychology, 430 Huntington Hall
Syracuse University, Syracuse, NY 13244
[email protected]
Introduction: There has been a great deal of controversy about whether the prevalence of autism is rising
(e.g., Shattuck, 2006; Wing & Potter, 2002 ). Evidence suggests, regardless of prevalence rates, that
children are being identified earlier than in years past (Mandell, Novak, & Zubritsky, 2005). Earlier identification may be due, in part, to better diagnostic tools that allow clinicians to detect symptoms in infants
(Landa & Garrett-Mayer, 2006) and toddlers (Charman & Baird, 2002; Mandell et al., 2005) and to the
growing public awareness of the disorder. When children are identified early they may be eligible to receive early intervention and/or preschool special education and related services. Such early interventions
may produce better outcomes for children (Levy, Kim, & Olive, 2006) and their families (Tonge et al., 2006)
than those children who do not receive support until school-aged.
Methods: The purpose of this study was to describe the child and family characteristics of infants, toddlers, and preschool-aged children diagnosed with autism spectrum disorders in one county in New York
State. Demographic characteristics of the child, family, age of diagnosis, and number and intensity of
services, were collected through a review of Health Department and Education archival records. Data
collection and analysis are ongoing.
Results/Discussion: Currently 124 children (birth – five) have been identified as diagnosed with an
autism spectrum disorder (autism, PDD-NOS). Of these children, the majority are preschool-aged (N=112)
and receiving special education and related services. Fewer diagnosed children are enrolled in the
county’s Early Intervention Program (N=12). Age at diagnosis, frequency/intensity of services, and race
and gender data will be reported on. Relative to the school-aged population of children in this county, there
is an estimated prevalence rate of 0.97%. Results will be discussed in light of the growing population of
children being served in Early Intervention and Preschool. Methodological issues pertaining to using
medical and educational records will be described.
Key References
Charman, T., & Baird, G. (2002). Practitioner review: Diagnosis of autism spectrum disorder in 2- and 3-year-old
children. Child Psychology and Psychiatry, 43, 289-305.
Landa, R., & Garrett-Mayer, E. (2006). Development in infants with autism spectrum disorders: A prospective study.
Journal of Child Psychology and Psychiatry, 47, 629-638.
Levy, S., Kim, A., & Olive, M. (2006). Interventions for young children with autism: A synthesis of the literature. Focus
on Autism & Other Developmental Disabilities, 21, 55-62.
Mandell, D. S., Novak, M. M., & Zubritsky, C. D. (2005). Factors associated with age of diagnosis among children with
autism spectrum disorders. Pediatrics, 116, 1480-1486.
Shattuck, P. T. (2006). The contribution of diagnostic substitution to the growing administrative prevalence of autism in
US Special Education. Pediatrics, 117, 1028-1037.
Tonge, B., Brereton, A., Kiomall, M., Mackinnon, A., King, N., & Rinehart, N. (2006). Effects on parental mental health
of an education and skills training program for parents of young children with autism: A randomized controlled
trial. Journal of the American Academy on Child Adolescent Psychiatry, 45, 561-569.
Wing, L., & Potter, D. (2002). The epidemiology of autistic spectrum disorders: Is the prevalence rising? Mental
Retardation and Developmental Disabilities Research Reviews, 8, 151-161.
133
15.
POSTER SESSION 2
Trajectory of Mathematics and Reading Skills in School-age Girls
with Fragile X Syndrome
Melissa M. Murphy & Michèle M. M. Mazzocco
Johns Hopkins University and Kennedy Krieger Institute
Contact: M Murphy 3825 Greenspring Ave Baltimore MD 21211
[email protected]
Introduction: Difficulty with math is documented among girls with fragile X syndrome as early as kindergarten
(Mazzocco, 2001) and persists through the elementary school years (Murphy & Mazzocco, in press). For
example, kindergarten girls with fragile X are distinguished from their peers on applied counting skills (e.g.,
identifying the Nth item in an array), but not rote counting skills (e.g., counting by ones). By 5th grade, girls
with fragile X differ from IQ-matched peers on untimed calculations and have less mastery of developmentally
appropriate math tasks, including counting. Reading ability may contribute to math as a mediator of math
performance, including word problem solving (Jordan et al., 2002). Relative to math, reading skills are a
strength for girls with fragile X. In early elementary school, girls with fragile X do not differ from IQ-matched
peers on nonword reading. In 5th grade, reading fluency and overall phonological access (a predictor of
reading) do not distinguish girls with fragile X from IQ-matched peers. Although there is crosssectional
support for the reported profile of math and reading-related skills, the developmental course of these skills
across the school years is unclear. The present study examines the trajectory of math and reading-related
skills in girls with fragile X during elementary and middle school, and the extent to which phonological access
predicts math performance.
Methods: Girls with fragile X who were seen at least two times in 1st-7th grade (n = 24) and a comparison
group of typically developing girls (n = 19), also seen during the same grades, were selected from a longitudinal
study. Girls with fragile X had lower IQ scores than the comparison group (p<.05), thus full scale IQ is
included as a predictor in the analyses. The Woodcock Johnson-Revised Calculation (available grades 1-6)
was used to assess math skills. Three rapid automatized naming (RAN) subtests, Colors, Numbers, and
Letters (available grades 1-7), assessed phonological access and retrieval.
Results: Growth curve analyses indicated that girls with fragile X had lower Calculation scores in 6th grade
(p<.01) and slower growth from 1st-6th grade (p<.05) than the comparison group. Relative to the comparison
group, girls with fragile X had longer response times on Color, Number, and Letter naming in 7th grade
(ps<.001), and their response times on Numbers between 1st-7th grade were decreasing at a faster rate
(p<.05). No group differences in growth were found for Colors or Letters. After controlling for FSIQ, differences
in response time on all three RAN tasks, but not Calculations, persisted. Numbers response time was a
significant predictor of 6th grade Calculation scores (p<.001); whereas FSIQ, and Colors and Numbers
response times predicted growth (ps<.05).
Discussion: The profile of math skills reported among young girls with fragile X continues to distinguish
them from their peers into middle school. Also, differences may emerge by middle school on reading-related
skills. Although data were not available on Calculations for the comparison group in 7th grade, examination of
7th grade scores for girls with fragile X suggests a plateau in performance in 7th grade. Group differences in
the trajectories of RAN subtests and the influence of FSIQ implicate the possible contribution of working
memory to RAN performance. Targeting intervention efforts in mathematics and phonological skills during
the elementary school years may be especially important.
References: Jordan et al. (2002). Achievement growth in children with learning difficulties in mathematics:
Findings of a two-year longitudinal study. JEP, 94, 586-597.; Mazzocco (2001). Math learning disability and
math LD subtypes: Evidence from studies of Turner syndrome, fragile X syndrome, and neurofibromatosis
Type 1. JLD, 34, 520-533.; Murphy & Mazzocco (in press). Mathematics learning disability in girls with fragile
X or Turner syndrome during late elementary school. JLD.
134
16.
POSTER SESSION 2
Lip Dynamics and Stereotypic Movement Disorders
Karl M. Newell, F. Aileen Costigan, Department of Kinesiology, The Pennsylvania State University and
James W. Bodfish, University of North Carolina, Chapel-Hill
This study examined the prevalence of stereotypic lip movements and their kinematic characteristics in
developmentally disabled adults (profound and severe mental retardation) that have been characterized
with stereotypic movement disorders (SMD), self-injurious behavior (SIB) and a contrast group that does
not meet the criteria for either of the other groups. The prevalence and kinematic characteristics of lip
stereotypies in persons with mental retardation that were diagnosed in different developmental disabilities
were investigated. The aim of the project was to examine the lip stereotypies as a function of different
developmental diagnoses as a way to provide information on the potential origins of stereotypies. A large
pool of participants were chosen from a developmental population and subsequently divided into one of
three groups based upon rating scale analysis of some key developmental repetitive disorders.
The participant pool included 118 adults, 76 males and 42 females, with mental retardation who resided at
a public residential facility. Their mean age was 45.05 years, with a standard deviation of 9.99 years. 16
of the participants functioned in the moderate range, 22 in the severe range, 79 in the profound range of
mental retardation (based on standardized behavior tests) and one participant was not rated. The etiology
of each participant’s mental retardation was unknown. The control group (Control) contained 35
participants and was defined as residents of the residential facility who did not meet criteria for the other
two groups. The stereotypy group (SMD) contained 58 participants and was defined as those individuals
who displayed 2 or more symptoms in all three categories on the DSM-IV Checklist or had a Childhood
Autism Rating Scale (CARS) score in the Autistic range or received a minimum score of 8 on the Behavior
Problems Inventory Stereotypy Subscale and a minimum score of 2 on the Stereotypy Checklist. The selfinjurious behavior group (SIB) contained 25 participants and included individuals who presented with self
injurious behaviors according to the Self Injury Trauma (SIT) Scale as administered by psychologists but
did not meet the criteria for the SMD group. The Control group consisted of 21 males and 14 females, the
STY group contained 6 males and one female, and the SIB group contained 1 male and 2 females.
The prevalence of lip stereotypies was proportionally higher in both the SMD and SIB groups than the
control group. Of the 118 participants, 12 individuals presented with visually identifiable stereotypic lip
movement that met the specific inclusion criteria for this study and were thus subjected to digitization of lip
dynamics and further analysis. Of these 12 selected participants, 8 were male and 4 were female.
Selected participants were divided by group as follows: 1 male belonged to the Control group, 6 males and
2 females belonged to the STY group, and 1 male and 2 females belonged to the SIB group. Kinematic
analysis of lip stereotypies on the participants that met the oscillatory criteria showed that they were more
constrained than in organization and at least variable as earlier data from a non DD control group. The
data provide further evidence of the enhanced prevalance of lip stereotypies in individuals with SMD and
for a different organization to the motor output than controls mimicking the stereotypy.
135
17.
POSTER SESSION 2
A Longitudinal Examination of the Mediating Role of Behavior Problems in Adaptation for
Families of Children With and Without Developmental Delays
Anita Pedersen y Arbona and Keith Crnic
Arizona State University
PO Box 871104, Tempe, AZ 85287-1104
[email protected], [email protected]
A family’s adaptation to a child’s developmental disability is a continuous process that evolves over time.
Although this process has attracted much theoretical and empirical debate, only a small number of studies
have explored family adaptation to a child’s disability from a longitudinal perspective. Early research
implied that families of children with developmental delays adapted poorly (Olshansky, 1962;
Wolfsenberger & Menolascino, 1970). Research has since shifted from highlighting the negative impact of
a child with delays to an exploration of the family’s positive and negative adaptation strategies (Baker,
Blacher, Kopp, & Kraemer, 1997). Currently, research suggests important associations among child developmental status, child behavior problems, and family adaptation. In particular, early behavior problems
may predict family adaptation more strongly than developmental status alone (Baker, Blacher, Crnic, &
Edelbrock, 2002). The proposed study will further explore the mechanisms that influence family adaptation
processes in a longitudinal study of young children and their families. Specifically, the proposed study aims
to determine the mediating role of child behavior problems in the link between child developmental level
and family adaptation over time.
Data for the proposed study will be drawn from a multi-site, longitudinal investigation which prospectively
examines the interrelations among children’s developmental status, family processes, child characteristics,
and the emergence of psychopathology in young children across the ages of 3 to 9 years. Data for the
study are collected using a multimethod, multi-informant approach involving structured parent interviews,
parent-completed questionnaires, and independent observations of parent-child interaction in naturalistic
and lab-based settings. Participants for the current study include 67 families of children with developmental delays (DD; Bayley MDI scores 75 or below) and 102 families of typically developing children (TD;
Bayley MDI score 85 or above). Developmental functioning was assessed at age 3. Level of child behavior
problems was obtained at age 6. Elements of family adaptation were measured at child age 6, and include
parent-reported questionnaire ratings of marital satisfaction, parent stress and parent psychological symptoms as well as naturalistic home observation ratings of dyadic parent-child and mother-father relationship
quality.
Preliminary analyses suggest that behavior problems (particularly externalizing problems) play a mediational role in the link between child developmental status and adaptation. Results indicate differential
prediction as a function of risk group. In families of children with developmental delays, parent stress,
parent psychological symptoms, and mother-child conflict are all significantly predicted by child externalizing problems. In families of typically developing children however, child externalizing problems significantly
predict only parent stress levels and parent psychological symptoms. These preliminary results suggest
more longitudinal exploration into the associations among child developmental level, behavior problems,
and family adaptation is warranted.
136
18.
POSTER SESSION 2
Cross-Syndrome Comparisons of Gesture Use inToddlers
Amy Philofsky, Ph.D., CCC-SLP, 1, Susan L. Hepburn, Ph.D. 1,
Deborah J. Fidler Ph.D.2, & Sally J. Rogers, Ph.D. 3
1
Department of Psychiatry, University of Colorado at Denver Health Science Center, 4200 E. 9th Avenue,
Box C268-30; Denver, CO 80262;
2
Human Development & Family Studies, Colorado State University;
3
MIND Institute, Department of Psychiatry, University of California, Davis;
*Contact: [email protected]
Interest in communicative gestures as precursors to language acquisition was originally fueled in the
1970’s. Bruner (1981) classified gestures that emerge during the first year of life in typical development
into three broad categories based upon the behavior’s function: 1) Behavioral Regulation (i.e., acts used
to regulate another’s behavior to obtain or restrict an environmental goal) 2) Social Interaction (i.e., acts
used to attract or keep another’s attention in order to associate) and 3) Joint Attention (i.e., acts used for
the purpose of directing another’s attention to share focus on an object or an event [Bruner, 1981]). Joint
attention, in particular, has been singled out as critical for further examination because it forms the
foundation for a number of later developing social and linguistic competencies (Carpenter & Tomasello,
2000; Charman et al., 2003; Mundy, Sigman, & Kasari, 2000; Sigman & Ruskin, 1999). Cross-syndrome
comparisons of the communicative gesture profile of young children have a number of clinical implications
and would provide for a better understanding of the differences and similarities between children with
different developmental disabilities.
Thus, the purpose of this study was to compare the communicative gesture profiles (i.e., Joint Attention,
Requesting—a form of Behavior Regulation, and Social Interaction) of children with different disabilities,
as well as typically-developing children, based upon the rate of gesture use occurring during
administration of the Early Social Communication Scales (ESCS; Siebert, Hogan & Mundy, 1982). 114
toddlers comprised six groups: Autistic disorder (AD; n = 28),Williams syndrome (WS; n = 14 ), fragile X
syndrome without co-morbid autism (FXS; n = 14 ), Down syndrome (DS; n = 18), Developmental Delay of
mixed etiology (DD; n = 14 ), and Typically developing (TD; n = 26). All groups were equated on nonverbal
mental age (NVMA) as measured by the Mullens Scales of Early Learning (MSEL), though the groups
were not equated on verbal mental age (VMA; ASD significantly lowest among clinical groups) or
chronological age (TD was significantly younger).
Results suggested significant mean group differences in Total Rate of Joint Attention [Kruskal-Wallis H (5,
114) = 28.8, p < .001], such that the AD and WMS groups performed similarly [U (1, 42) = 166, p = .42]
and significantly worse than all of the other 4 groups. In terms of rate of requesting behaviors, significant
mean group differences in Total Rate of Requesting were also observed [Kruskal-Wallis H (5, 113) = 25.6,
p < .001] such that the AD group performed significantly worse on rates of requesting behaviors than 4
other groups (not DS) and TD performed significantly better than DS and FXS. In terms of social
interaction behaviors, mean group differences in Total Rate of Social Initiations were noted [Kruskal-Wallis
H (5, 103) = 22.4, p < .001] such that the AD group performed significantly worse than all of the other 5
groups in total rate of social initiations. Implications are discussed for language development and
treatment, as well as the importance of cross-syndrome comparisons for elucidating early emerging
behavioral phenotypes.
This research was supported by the NIH (U19 HD35468-07) and the March of Dimes.
137
19.
POSTER SESSION 2
Down Syndrome, and Congenital Heart Defect: Maternal Experiences of Bereavement
Deirdre Reilly, Frances Vaughan, and Jaci Huws
School of Psychology, University of Wales, Bangor.
Contact: [email protected]
Background: Congenital heart defect (CHD) has been reported in almost 50% of newborns with Down
syndrome (Frid et al, 2002). Children with Down syndrome and CHD have been shown to be at higher risk
of mortality than children with Down syndrome without CHD. CHD is also one of the most often reported
medical disorders on death certificates of adults with Down syndrome, particularly between the ages of 20
and 29 (Yang, Rasmussen, & Friedman, 1999). This study aimed to investigate the bereavement
experiences across the life span of mothers who lose a child with Down syndrome and CHD.
Method: 31 bereaved mothers of children with Down syndrome and a CHD were recruited through a
support group in the UK. Average time since loss was 10.3 years (SD = 6.61: range 1 – 23). Mothers
completed measures of grief, coping, anxiety and depression, and a retrospective measure of their
positive perceptions of parenting a child with intellectual disability.
Results: Overall 56.2 % and 28.2% of mothers scored in the possible or probable range for clinical
anxiety or depression disorders respectively, which is high compared to the UK population in general.
Revised Grief Experienced Inventory (RGEI) scores were similar to previously reported scores for
bereaved parents (M = 74.48; SD = 30.79). Mothers reporting higher levels of grief tended to report more
positive perceptions of their experience of raising and losing a child with Down syndrome (r = .511; p =
.003). Also mothers with higher grief tended to use more active avoidant (r = .475; p = .006) coping
strategies through their bereavement, but use of problem focused coping strategies was not related to
grief.
Discussion: As in the bereavement literature more generally, avoidant coping seems to be maladaptive in
relation to mothers’ grief experiences. The association between current grief and positive perceptions of
the child with Down syndrome requires further exploration but may have important implications for
supporting parents through bereavement.
Key References
Yang, Q., Rasmussen, S. A., & Friedman, J. M. (2002). Mortality associated with Down’s syndrome in the
USA from 1983 to 1997: a population-based study. The Lancet, 359(9311), 1019-1025.
Frid, C., Drott, P., Lundell, B., Rasmussen, F., & Anneren, G. (1999). Mortality in Down Syndrome in
relation to congenital malformations. Journal of intellectual disability research, 43(3), 234-241.
138
20.
POSTER SESSION 2
Correlations between Locus of Control and Assertiveness and Knowledge of Abuse and
Decision-Making Performance among Individuals with Moderate to Severe Disabilities
Anne Spillane
National University
910 Stratford Court, Del Mar, CA, 92014
Contact: [email protected]
Introduction: Individuals with intellectual disabilities (ID) have been shown to be at increased risk for abuse
(Sobsey, 1994). The present study investigated correlations between locus of control and assertiveness and
knowledge of abuse and decision-making performance in response to interpersonal situations involving abuse
among individuals with more severe ID.
Method: This study investigated correlations between the locus of control and assertiveness of 40
adolescents with moderate to severe ID, and their knowledge of abuse and decision-making performance
in response to interpersonal situations involving abuse.
Results: Participants with a more external locus of control were less able to identify situations of abuse
and the type of abuse (sexual abuse vs. physical abuse vs. verbal abuse). Participants who were more
assertive were more able to identify situations of abuse and the type of abuse (sexual abuse vs. physical
abuse vs. verbal abuse). Participants who were more assertive were also more likely to give an
independent self-protective response on the multiple choice questions on the self decision-making scale.
Discussion: Previous studies have demonstrated that locus of control is linked to attending to information
and the environment (Lefcourt, 1976). An external locus of control may have caused these participants to
be less attentive to the salient information needed to identify situations of abuse and to differentiate
between the types of abuse. Motivational aspects of personality, such as assertiveness, have been
discussed as having an influence on the performance of individuals with mental retardation (Zigler, 1999).
The correlation found here between assertiveness and knowledge of abuse further strengthens the
argument for the connection between motivational and personality factors and performance. Finally,
motivational and control factors have been recognized as being an important component of deciding upon
a course of action (Kuhl, 1986). Individuals who are more assertive may have the necessary confidence
to choose an independent course of action, instead of relying on others to act for them.
Key References:
Kuhl, J. (1986). Motivation and information processing: A new look at decision making, dynamic change,
and action control. In R.M. Sorrentino & E.T. Higgins (Eds.) Handbook of motivation and cognition:
Foundations of social behavior. (pp. 404-434). New York: Guilford Press.
Lefcourt, H.M. (1976). Locus of Control: Current Trends in Theory and Research. Hillsdale, NJ:
Lawrence Erlebaum and Associates.
Sobsey, D. (1994). Violence and Abuse in the Lives of People with Disabilities. Baltimore: Paul H. Brookes.
Zigler, E. (1999). The individual with mental retardation as a whole person. In E. Zigler & D. BennettGates (Eds.) Personality Development in Individuals with Mental Retardation. Cambridge:
Cambridge University Press.
139
21.
POSTER SESSION 2
Sibling Relationship Quality for Individuals with Mental Retardation:
A Comparison of Sibling Reports
Shana Strickland, Vivian Piazza, & Frank J. Floyd
Georgia State University
Contact: Shana Strickland Georgia State University
PO Box 5010 Atlanta, GA 30302
Contact: [email protected]
Much of the research examining sibling relationships for individuals with mental retardation (MR) has relied
on reports from either the sibling without disabilities or a parent. To date, studies have rarely considered
perceptions of the relationship by the individual with MR. In order to better address how sibling
relationships might affect individuals with MR, it is important to take into consideration their perceptions of
these relationships. Further, most studies have examined sibling relationship quality only in childhood. In
order to address these gaps in the research, the present study examined evaluations of sibling
relationships by adolescents and young adults with MR, and compared their evaluations to measures of
relationship quality completed by the siblings.
Participants were 29 adolescents and young adults with mild or moderate MR and their siblings. A target
sibling was randomly selected for those participants who had more than one sibling. Individuals with MR
ranged from age 14 to 30 years (M = 21.79), and their siblings were aged 13 to 31 years (M = 20.34). The
individuals with MR completed the My Family and Friends (Reid & Ramey, 1992) measure during a faceto-face interview, which required them to rate their perceptions of the quality of emotional support from the
sibling. Specific items focused on satisfaction with discussing feelings and perceptions of the sibling’s level
of understanding. Siblings completed the Sibling Relationship Questionnaire (SRQ, Furman & Buhrmester,
1985), which measured multiple domains of the sibling relationship. To examine similar domains for both
siblings’ reports, we focused on the Warmth/Closeness scale of the SRQ, along with the subdomains of
this scale.
Initial analyses indicated that the individuals with MR generally perceived the quality of emotional support
from their siblings positively. For example, on a 6 point scale, all individuals reported that they felt at least
“a very little bit better” (rating = 2) after sharing feelings with their siblings. On the SRQ, the siblings rated
the relationship highest in the areas of affection and admiration by their brother or sister with MR, and
lowest in the areas of similarity and intimacy between the siblings. Correlations revealed that quality of
emotional support as rated by the individuals with MR was significantly positively associated with the
siblings’ total scores for Warmth/Closeness, and with the subdomains prosocial, affection, and admiration
by their brother or sister. However, the evaluations by the individuals with MR were not significantly
correlated with the siblings’ ratings of companionship, similarity, intimacy, and admiration by the sibling.
Overall, the results revealed that evaluations across siblings were generally consistent regarding several
positive aspects of the sibling relationship. However, inconsistencies that occurred might emerge from the
asymmetrical nature of these relationships with, for instance, highly supportive siblings unlikely to perceive
reciprocity in terms of high levels of companionship, similarity, or intimacy in the relationship. Also,
relatively low levels of similarity and intimacy might be a function of the ages of the siblings, wherein a
general decline in contact typically is seen in adolescence and young adulthood. Thus, the results highlight
the importance of re-interpreting certain relationship constructs when working with individuals with MR.
140
22.
POSTER SESSION 2
Examining Contingency Control Deficits in Children with and without Autism
Louis P. Hagopian; The Johns Hopkins University School of Medicine
Lisa M. Toole; Kennedy Krieger Institute and University of Maryland, Baltimore County
Eric Boelter; Kennedy Krieger Institute
Heather K. Jennett; Kennedy Krieger Institute
Gregory A. Lieving; West Virginia University Institute of Technology
Keith J. MacWhorter; Kennedy Krieger Institute
Elizabeth L. Tobin; Towson University
Autism is characterized by deficits in communication and social skills, and restricted, repetitive, and
stereotyped behavior patterns (DSM-IV, 1994). These deficits can be conceptualized in terms of global
deficits in contingency control, defined broadly as the ability to adapt and change behavior in accordance
with contingencies. Deficits in contingency control would result in the persistence of behavior after it is no
longer functional; decreased adaptability of behavior to new environments or changes in familiar
environments; and patterns of behavior that show little variation in form and may be repetitive or
stereotypic. In the current studies, we sought to examine the phenomena of behavioral sensitivity related
to contingency control, specifically, persistence of responding and decreased behavioral variability in both
topography of response and rate.
In the first study, participants earned brief access to reinforcement for responding on a touch-screen
monitor. In the response acquisition phase, stimuli were present on the screen and reinforcement was
available contingent on a 3-touch sequence. In the extinction phase, no reinforcement was provided for
responding, and sessions were conducted until responding was at a rate of less than 1 response per
minute or until 12 sessions had been conducted. Preliminary results suggest that responding by the
participants with autism is characterized by greater resistance to extinction when compared to control
children (i.e., participants without a diagnosis of autism). In the second study, we compared average
break points and overall local response rate of responding on progressive ratio schedules. Again,
participants earned brief access to reinforcement for responding on a touch-screen monitor. During the
Progressive Ratio Phase, response requirements were systematically increased until termination criteria
were met. Preliminary results suggest that participants with autism exhibited greater persistence,
characterized by slower, steady rates with shorter post-reinforcement pauses and higher break points.
CONTACT INFORMATION: Lisa M. Toole, M.A., BCBA
Kennedy Krieger Institute
707 N. Broadway
Baltimore, MD 21205
[email protected]
141
23.
POSTER SESSION 2
Understanding of the Causes of Emotions in Others by Children with Down’s Syndrome
K.R. Williams1, J.G. Wishart1, D.S. Willis2, & T.K. Pitcairn1
1
University of Edinburgh; 2 Napier University
Katie Williams
Moray House School of Education
University of Edinburgh, Holyrood Road
Edinburgh EH8 8AQ, Scotland, UK
[email protected]
Introduction: Previous research has suggested that some children with Down’s syndrome experience
difficulties in recognition of emotional expressions (Kasari, Freeman, & Hughes, 2001; Williams et al.,
2005; Wishart & Pitcairn, 2000), but relatively little work has explored their emotional understanding more
broadly. This study investigated children’s understanding of the causes of emotions in others, using a
cartoon task that controlled for story-comprehension ability and general task demands by comparing
children’s ability to identify the antecedents of physical events versus emotional events.
Methods: Three child groups matched on mental age participated in this study: 21 children with Down’s
syndrome and 21 with non-specific intellectual disability aged 9 – 19 years, plus 21 typically developing
children aged 3 – 6 years. The groups did not differ significantly on language comprehension or emotion
labelling ability. Children were given 20 three-part cartoon stories: 4 depicted physical events, 8 depicted
emotions caused by a physical action, and 8 depicted emotions caused by a person. The emotions
included were happiness, sadness, surprise and fear. For each story the first and last picture were
presented and the children asked to select the correct middle picture from a choice of three. Children were
then asked to tell the story.
Results: Groups did not differ significantly in their ability to select the correct antecedents in any condition.
However, analysis of individual emotions suggested that the children with Down’s syndrome had
somewhat more difficulty than the typically developing children in identifying antecedents to expressions of
fear. Ongoing analysis of the content of children’s stories may reveal further subtle differences in their
understanding of emotion causality.
Discussion: Findings from this study support previous research that has suggested that emotional
understanding may be an area of difficulty for some children with Down’s syndrome. However, these
difficulties may be very subtle and detectable only in comparison with typically developing children,
presenting challenges both to theories of emotion recognition and behaviour phenotype theory.
Key references:
Kasari, C., Freeman, S.F.N., & Hughes, M.A. (2001). Emotion recognition by children with Down
syndrome. American Journal on Mental Retardation, 106, 59-72.
Williams, K.R., Wishart, J.G., Pitcairn, T.K., & Willis, D.S. (2005). Emotion recognition by children with
Down syndrome: investigation of specific impairments and error patterns. American Journal on Mental
Retardation, 110, 378-392.
Wishart, J. G. & Pitcairn, T. K. (2000). Recognition of identity and expression in faces by children with
Down syndrome. American Journal on Mental Retardation, 105, 466–479.
142
24.
POSTER SESSION 2
Imitation in Young Children with Autism and Other Developmental Delays
Connie S. Wong
Cleveland State University, College of Education and Human Services
2121 Euclid Avenue, Rhodes Tower 1325
Cleveland, OH 44115-2214
[email protected]
Young children with autism have difficulty imitating other people’s actions. Impairments in imitation ability
could negatively affect children’s social-emotional development, a specific area of concern for children
with autism. Research is ongoing to determine the mechanism or deficit underlying imitation performance
in children with autism. The objective of this study was to explore child characteristics and behaviors that
may influence children’s performance on imitation tasks.
Fifty-five children were recruited from a suburban special-education public preschool. In this study,
participants were analyzed in two groups: children with a clinical diagnosis of autism (N=27) and children
with other developmental delays (N=28). The children ranged in age from three to five years old and their
mental ages ranged from 18 to 59 months. Child characteristics and demographics were not significantly
different between the two groups. In this study, children were assessed using nine items from the Imitation
Battery (Rogers, Hepburn, Stackhouse, & Wehner, 2003), which included 3 manual acts, 3 actions on
objects, and 3 oral-facial movements. Additionally, children were also assessed on measures of joint
attention and symbolic play.
Consistent with existing research, results indicate that compared to children with other developmental
delays, children with autism completed fewer imitation tasks correctly. However, a closer examination of
child characteristics using analysis of variance procedures showed significant main effects of
developmental scores as well as a significant interaction effect between disability group and
developmental scores. While children with higher mental and language ages generally completed more
imitation tasks successfully than children with lower developmental abilities, children with autism showed a
much greater disparity. Whereas children with higher developmental scores in both groups performed at
similar levels, children with lower developmental scores in the autism group failed significantly more
imitation tasks than those children with other developmental delays. In addition, while preliminary results
did not find significant groups differences in reversal errors and errors of style, results did indicate that
children with autism were less engaged with the experimenter and displayed fewer coordinated joint looks
than the children with other developmental delays during the imitation battery. Finally, performance on the
imitation tasks was not related to other measures of joint attention or symbolic play. These findings
suggest the importance of cognitive and language abilities in the development of imitation skills.
Rogers, S.J., Hepburn, S.L., Stackhouse, T., & Wehner, E. (2003). Imitation performance in toddlers with
autism and those with other developmental disorders. Journal of Child Psychology and Psychiatry,
44, 763-781.
Williams, J.H.G., Whiten, A., & Singh, T. (2004). A systematic review of action imitation in autistic spectrum
disorder. Journal of Autism and Developmental Disorders, 34, 285-299.
143
25.
The Relationship Among Vocabulary Knowledge, Phonological Awareness, and
Reading Performance in Children with Mild Intellectual Disabilities
Rose A. Sevcik, Justin C. Wise and Robin D. Morris
Georgia State University, Dept. of Psychology, PO Box 5010, Atlanta, GA 30302-5010
[email protected]
Limited research exists concerning the literacy skills of children with intellectual disabilities. Children with
intellectual disabilities primarily have been taught to read through sight words since it has been assumed
that individuals with intellectual difficulties can not benefit from phonemic instruction due to limited
cognitive skills and associated language difficulties. Recent studies, however, suggest that phonological
awareness is related to reading performance (e.g., Gombert, 2002; Snowling, Hulme, & Mercer, 2002) and
that they can benefit from phonics-based literacy instruction (e.g., Cupples & Iaocono, 2002).
In our program of research, a multidimensional theoretical model of developmental reading processes has
been used to identify critical instructional components (decoding, fluency, and text comprehension)
needed to acquire reading skills. These instructional programs are based on converging evidence, from a
range of learners, that early reading incorporates two areas of linguistic skill: phonological awareness and
naming retrieval/access speed (Wolf & Bowers, 1999). The impact of the reading programs on both early
developing reading skills (e.g., phonological awareness, word decoding) and the development of fluency
and comprehension skills, along with other related language skills are examined. Using a randomized
experimental design, reading instruction was delivered in small groups (n=4) taught by highly experienced
teachers. In this poster, the relationships between vocabulary knowledge, phonological awareness skills,
and word and nonword identification performance are examined. Data reported here are from the
baseline time point collected during the first year of the project.
Forty-seven students classified by their school district as eligible for services for students with mild intellectual disabilities participated. The sample consisted of 30 male and 17 female students with an ethnic
representation of 24 Caucasian, 16 African American, 4 Hispanic, and 3 Asian students. Their mean
language age in months assessed by the PPVT-III was 66.09 (range = 21-119), and their mean chronological age in months was 112.57 (range = 84-142). The etiology of the intellectual impairments evidenced
by these students was very heterogeneous and included Down syndrome, Fragile X syndrome, and etiology unknown.
Prior to receiving instruction, students were administered a comprehensive battery of linguistic and reading-related measures. Student performance on measures of receptive and expressive vocabulary, measures of phonological awareness, and measures of word and nonword identification were included for
these analyses. For all analyses, raw scores were used to avoid floor effects and to increase variability
among scores.
Partial correlation analyses controlling for age indicated that measures of phonological awareness correlated significantly (p < .05) with measures of word (r = .38-.63) and nonword (r = .40-.45) identification.
With respect to vocabulary knowledge, the only significant correlation was between receptive vocabulary
knowledge and word identification performance, r = .38, p < .05). Hierarchical regression analyses indicated that after controlling for age and vocabulary knowledge, the ability to blend phonemes to produce a
word accounted for a large and significant amount of unique variance of both word (47%), F (1,42) =
16.27, p < .001, and non word (26%), F(1,42) = 7.53, p < .01, identification.
Results of these analyses suggest that phonological awareness skills are significantly correlated with
reading performance for children with intellectual disabilities. Further, hierarchical regression analyses
indicated that this group of children relied heavily on phonological awareness skills for the purpose of word
and nonword identification. Implications of this study are that children with mild intellectual disabilities can
learn to read similarly to typical children and suggest that these children can benefit from phonics-based
literacy instruction.
144
S y m p o s i u m 10
S Y M P O S I U M
10
Individual Differences in Adaptation in Parents and Siblings
of Children with ID – Process Mechanisms and Implications
for Support
Chair:
Malin B. Olsson
Department of Psychology, Goteborg University
145
SYMPOSIUM 10
Individual Differences in Adaptation in Parents and Siblings of Children with ID – Process
Mechanisms and Implications for Support
Chair: Malin B Olsson, Ph.D.
Department of Psychology, Goteborg University
Longitudinal Perspectives on Sibling Impact: The Role of Clinical Behavior Problems
Jan Blacher & Cameron Neece,
UCLA, Graduate School of Education
University of California
Acceptance as a Longitudinal Predictor of Stress, Anxiety and Depression in Mothers of
Children with Intellectual Disabilities
Tracey Lloyd & Richard Hastings
University of Wales Bangor
Family Process in Families of Young Children with Intellectual Disabilities: Depression and
Inter-Parental Conflict
Zolinda Stoneman
Institute on Human Development and Disability/UCEDD,University of Georgia
The Impact of Psychological Variables on Well-Being of Parents of Children with ID
Malin B. Olsson
Department of Psychology, Goteborg University
146
SYMPOSIUM 10
Longitudinal Perspectives on Sibling iImpact: The Role of Clinical Behavior Problems
Jan Blacher (Graduate School of Education, UC Riverside)
[email protected]
Cameron Neece (Dept. of Psychology, UCLA) Los Angeles, CA 90095
Introduction: The study of siblings of children with developmental disabilities (DD) is fraught with layers of
complexity. Methodological and conceptual issues not withstanding, (Hodapp, Glidden, & Kaiser, 2005;
Stoneman, 2005), there are opportunities for examining the impact of a child with DD from a number of
perspectives. This study involves both mother and father reports, taken longitudinally, of sibling impact.
Specifically, we were interested in parent perceptions of sibling impact (when the target child was DD or
typically developing, TD), when controlling for concomitant target child behavior problems, and when
considered over time.
Method: We followed n=98 children with borderline to moderate intellectual disability (ID) and n=144
typically developing children (TD) from child age 3 to 5 years. Overall, the child sample was 57% boys
and 61% Caucasian. The parent sample was 86% married and 51% of mothers and fathers were college
graduates. The SES variable, mother’s education, was lower in the ID group, so this was co-varied in all
status group comparisons. At child ages 3, 4, and 5 both mothers and fathers completed two measures of
interest in this study: the Family Impact Questionnaire, FIQ (Donenberg & Baker, 1993) to assess stress,
and the Child Behavior Checklist, CBCL (Achenbach, 2001) to assess the extent and clinical nature of
child behavior problems. There are six subscales of the FIQ, but for this study the focus was on the impact
of the target child on non-disabled siblings.
Results: First, we looked at parental ratings of sibling impact at age 3. In all cases, both mothers and
fathers of children with DD rated siblings as experiencing higher negative impact than did parents of TD
target children. Second, we examined the sibling impact score across ages 3, 4 and 5 (in a repeated
measures ANOVA), with a significant status group difference. The ANOVA revealed no significant time
effect, indicating that this phenomenon is stable. There was a significant effect of the covariate (mother’s
grade in school); mothers with a higher education level perceived less negative impact. The same status
group effect was reported by fathers; however, there was no effect of the continuous covariate of grade in
school for fathers. Third, in another repeated measures ANOVA using mother data, with between subject
factors child status (TD or DD) and behavior problems (clinical or not), status group differences were no
longer significant; there was no time effect. There was a highly significant behavior problems effect and a
significant status by behavior problem interaction. An examination of means revealed that the lowest mean
at each timepoint was for the TD children with non-clinical problems (as expected). However, the highest
mean at each timepoint was for the TD children with clinical behavior problems – higher than for the
children with ID and behavior problems! In other words, the presence or absence of serious behavior
problems made more of a difference in TD families.
Discussion: Results reported here support earlier reports from this project (see Baker et al, 2002, 2003)
that implicate child behavior problems as having more negative impact on families than the designation of
developmental delay per se. In this study, we examined the impact of the target child (ID or TD) on nondisabled siblings, and again, when controlling for clinical level behavior problems, status group differences
disappear. These findings have implications for those studying stress in families of specific syndrome
groups, as the presence or absence of clinically significant behavior problems are likely to have greater
negative impact than syndrome designation per se. Fortunately for family stress levels, many behavior
problems – regardless of etiology — are amendable to intervention.
147
SYMPOSIUM 10
Acceptance as a Longitudinal Predictor of Stress, Anxiety and Depression in
Mothers of Children with Intellectual Disabilities
Tracey Lloyd and Richard P. Hastings
University of Wales, Bangor
Contact: Tracey Lloyd, University of Wales Bangor, Brigantia Building,
Penrallt Road, Bangor, Gwynedd, UK, LL57 2DG.
[email protected]
Introduction: As with psychological process variables generally, there has been little research on
psychological acceptance as a predictor of adjustment in family members of people with intellectual
disabilities. In cross-sectional research, we showed previously that acceptance was a significant predictor
of maternal anxiety and depression, and marginally of stress. In the present paper, we explore data
addressing these relationships over time.
Methods: Fifty seven mothers of children attending special schools completed questionnaire surveys 18
months apart. Data were gathered using the Acceptance and Action Questionnaire (AAQ) to measure
acceptance of unwanted thoughts/feelings, the Hospital Anxiety and Depression Scale (HADS) measuring
mental health symptoms and the Questionnaire on Resources and Stress (Short Form) (QRS-F) as an
overall measure of family stress.
Results: Acceptance was found to be modestly stable over 18 months (r = .603). For anxiety, the change
over 18 months in acceptance predicted Time 2 scores after Time 1 scores were controlled. A similar
pattern of results was found for the prediction of depression at Time 2. For anxiety and depression,
change in these variables over time also predicted Time 2 acceptance after controlling Time 1 acceptance.
After controlling for other variables (notably, child behaviour problems), stress and acceptance were not
related over time.
Discussion: These data suggest a bidirectional relationship over time between acceptance and mental
health symptoms in mothers of children with intellectual disabilities. Interventions that directly address
acceptance as a process variable (e.g. Acceptance and Commitment Therapy) may be useful in the
support of parents with a child with intellectual disabilities.
148
SYMPOSIUM 10
Family Process in Families of Young Children with Intellectual Disabilities:
Depression and Inter-Parental Conflict
Zolinda Stoneman,
Institute on Human Development and Disability/UCEDD, University of Georgia,
850 College Station Rd., Athens, GA, 30602
[email protected]
It is important to understand the processes that lead to wellness or compromised functioning within
families. This study examined the associations between depression in mothers and fathers of young
children with intellectual disabilities and inter-parental conflict. It was hypothesized that for both mothers
and fathers, elevated levels of self-reported depression would be associated with economic stress and
inter-parental conflict and that the negative influences of these variables would be buffered by the
presence of satisfying social support.
Forty-five married couples participated in the study. Families had one child with an intellectual disability
between the ages of 2 and 6 years who was living at home. Approximately half of the children were male
and half female. Slightly less than half of the children had Down syndrome. Families were 80% EuroAmerican and 20% African-American. Families were recruited from early intervention programs and parent
organizations. Mothers and fathers independently completed a demographic information form, the Center
for Epidemiologic Studies Depression Scale (CES-D), the O’Leary-Porter Inter-Parental Conflict Scale,
and the Dunst Family Support Scale which measures satisfaction with various sources of social support.
Initial analyses were conducted to examine demographic predictors of parent depression. For mothers,
depression was correlated with less family income and lower maternal education. For fathers, depression
was related to lower family income. Child competence measures (obtained from the Battelle) were
unrelated to depression in either parent. There were positive correlations between mothers’ and fathers’
reports of depression, inter-parental conflict, and satisfaction with social support. Paired samples t-tests
revealed no differences between mothers and fathers on their reports of depression, conflict, or social
support.
The study hypotheses were tested using hierarchical regression models. For mothers, lower incomes and
higher levels of inter-parental conflict predicted heightened levels of depression. For fathers, lower
income was the only predictor of self-reported depression. Satisfaction with social support did not
evidence the expected buffering effect. The pattern of findings was quite different for mothers and fathers.
Discussion of study findings will focus on the importance of parent gender in family research.
149
SYMPOSIUM 10
The Impact of Psychological Variables on Well-Being of Parents of Children with ID
Malin B Olsson
Department of Psychology, Goteborg University,
Box 500, 405 30, Göteborg, SWEDEN,
e-mail: [email protected]
Several studies have reported that mothers of children with ID are more likely to show signs of
psychological distress than mothers of children without ID (Olsson & Hwang, 2001). But the individual
variation is substantial; more than 50% of the parents of children with ID show no signs of depression.
Since most studies have only measured the negative end of the well-being continuum (depression or
stress) we know very little about factors that differentiate parents who experience poor well-being from
those who experience positive well-being. The present study investigated the relation between
psychological differences in parents (adult attachment style, sense of coherence and perceived control)
and parental variations in well-being.
Methods: The participants were approximately 75 families of children with ID (approximately 75 mothers
and 50 fathers).The children were from 0 to 6 years of age, and had been identified as having ID during
the last six months. The mean child age was 3 years and 3 months, a majority was boys.
Measures:
Well-being
BDI-2r (which actually is BDI with 3 positive alternatives added to each of the 21items (Chow &
Brenton,2000).
Socioeconomic situation
Hardship questionnaire (12 items adapted after Emerson at al., 2004). Income more than 2000 € /
month
Child characteristics
Development (5 items rated in relation to peers)
Behavior problems (SDQ (15 selected items), Goodman, 1999) & Nisonger scale (10 selected i
tems)(Aman, Tassé, Rojhn & Hammer, 1995)
Psychological factors
Perceived parental control (11 items, Hagekull & Bohlin, 2002)
Sense of coherence, short form (13 items, Antonovsky, 1987)
The Relationship Questionnaire (RQ) (Bartholomew & Horowitz, 1991)
Results: Preliminary results suggest that high and low risk parents (in terms of child behavior problems
and SES) differ in level of well-being and that psychological factors (in terms of SoC, secure adult
attachment style and perceived control) mediate the relationship.
Discussion: No single variable can explain the variance in well-being, well-being is dependant on the
complex interplay of risk and protective factors. In clinical work we need to work toward diminishing the
risk factors and boosting the protective factors.
Key References:
Antonovsky, A. (1987) Unraveling the mystery of health. How people mange stress and stay well. San Fransisco, Cal.
Jossey-Bass
Bartholomew, K. & Horowitz, L. M. (1991). Attachment styles among young adults: A test of a four-category model.
Journal of Personality and Social Psychology, 61, 226-244.
150
S y m p o s i u m 11
S Y M P O S I U M
11
Phenotypic Responses to Communication Intervention by
Young Children with Down Syndrome
Chair:
Steven F. Warren
Institute for Life Span Studies, University of Kansas
Discussant: Len Abbeduto
Waisman Center, University of Wisconsin-Madison
151
SYMPOSIUM 11
Phenotypic Responses to Communication Intervention by
Young Children with Down Syndrome
Chair:
Steven F. Warren
Institute for Life Span Studies, University of Kansas
Discussant: Len Abbeduto
Waisman Center, University of Wisconsin-Madison
Treatment (non)Effects and Predictors of Communication and Language Development in
Children with Down Syndrome
Paul J. Yoder1 and Steven F. Warren2
1
John F. Kennedy Center for Human Development, Vanderbilt University
2
Institute for Life Span Studies, University of Kansas, Lawrence
Response by Children with Down Syndrome to Prelinguistic Milieu Teaching
Nancy C. Brady1, Steven F. Warren1, Marc E. Fey2 Lizbeth H. Finestack2, Shelley L. Bredin-Oja2, &
Kandace K. Fleming1
1
Institute for Life Span Studies, University of Kansas, Lawrence
2
University of Kansas Medical Center, Kansas City, KS
Effects of Early Augmented Language Intervention on the Communication Skills of Young
Children with and without Down Syndrome
Mary Ann Romski, Rose A. Sevcik, Lauren B. Adamson, Melissa Cheslock, Ashlyn Smith, Mike
Barker, and Roger Bakeman
Georgia State University, Atlanta, GA
152
SYMPOSIUM 11
Treatment (non)Effects and Predictors of Communication and Language
Development in Children with Down Syndrome.
Paul J. Yoder1 and Steven F. Warren2
Vanderbilt University, Peabody 328, Nashville, TN 37203, [email protected]
2
Life Span Institute, 1000 Sunnyside Dr., Rm. 1052, Lawrence, KS. 66045-7555
1
Introduction: It is important to maximize the developmental trajectories of children with intellectual
disabilities. Understanding the sources of variance in these trajectories may eventually improve our ability
to do so. The goal of this presentation is to shed light on the sources of variance in the growth of
communication and language in children with Down syndrome (DS) in response to intervention.
Methods: This presentation is focused on the results of two sets of analyses from a study of 39 initially
prelinguistic toddlers with intellectual disabilities and their parents. Seventeen of these children had DS.
The first set of analyses utilized a randomized group experiment (treatment vs control) with extensive pretreatment measurement of parent-child and child characteristics, one of which was etiology (DS vs mixed
etiology). The treatment of interest was Responsive Education and Prelinguistic Milieu Teaching (RE/
PMT). The key outcome of interest was frequency child-initiated requests in semi-structured parent-child
sessions. The second set of analyses utilizes a longitudinal correlational design to identify the unique
predictors of later language development in children with and without DS matched on IQ, age, and
vocabulary level at Time 1. In these analyses, “Time 1” is used to identify measures derived immediately
after the treatment phase ended. The “Time 2” language outcome was lexical density (number of different
words used in a communication sample) and was measured 6 months after the treatment phase had been
completed. The treatment group assignment did not affect which Time 1 predictors accounted for variance
in later language. Time 1 predictors of later language were identified after controlling for Time 1 language.
Etiology main effects and etiology by Time 1 predictor interactions predicting later language were also
tested. Key Time 1 predictors were frequency of prelinguistic comments, frequency of vocal
communication with consonants, frequency of parental optimal responding (vocal imitation, linguistic
mapping, and compliance).
Results: The findings of the first set of analyses indicated that RE/PMT affected frequency of requesting
in children without DS, but did not do so in children with DS. To our knowledge, this is the only
demonstration that DS, per se, affected the efficacy of a communication treatment. The results of the
second set of analyses indicated that children DS had much lower language levels (d = 1.46) and much
less variance in language than matched children without DS. Optimal parental responsivity predicted later
lexical density, regardless of etiology (R square change = .20). Frequency of vocal communication with
consonants (R square change for product term = .18) and frequency of commenting (R square change for
product term = .12) predicted later lexical density only in children without DS.
Discussion: The results support the conclusion that children with DS have unique problems acquiring
language and responding to some language intervention techniques. The presentation offers speculations
as to why young children with DS did not respond to RE/PMT in a manner similar to other children with
developmental delays.
This research was supported by NICHD grants R01 HD34520 and P30 HD15052
153
SYMPOSIUM 11
Response by Children with Down Syndrome to Prelinguistic Milieu Teaching
Nancy C. Brady1, Steven F. Warren1, Marc E. Fey2, Lizbeth H. Finestack2, Shelley L. Bredin-Oja2,
& Kandace K. Fleming1
1
Life Span Institute, 1000 Sunnyside Ave, Rm. 1052, Lawrence, KS. 66045
2
University of Kansas Medical Center, Kansas City, KS.
Introduction: Prelinguistic milieu teaching is a well-researched intervention that has been implemented
with children with a variety of developmental delays, including Down syndrome (DS). Child communication
targets include prelinguistic forms such as gestures and non-word vocalizations that are clearly directed to
a partner and are used to request, comment and initiate social interactions. To optimize the effects of this
intervention, parents and other caregivers are taught ways to support and respond to child communication
attempts. The resulting intervention has been termed RE/PMT.
Methods: Fifty-one children with developmental disabilities, age 24-33 months, with no more than 10
expressive words or signs, were randomly assigned to treatment/no treatment group. Thirteen children in
each group had DS. The intervention component lasted six months. During this time, parents received 8
individual one-hour lessons on responsive interaction strategies, and children received PMT in their homes
or classrooms 4 days per week, in 20 minute sessions.
Based on earlier findings of Yoder and Warren (2002) PMT procedures were modified to make them more
effective with the children with DS. These changes included limiting the use of certain kinds of prompts.
Teachers observed that the children with DS, in particular, were not likely to respond to prompts such as
“show me” or “tell me,” or “do this.” Therefore interventionists paused and waited for responses, provided
full models if the response was not demonstrated during this pause, and then went on with the activity. In
addition, teachers initially accepted approximations of target responses. Breaking down the target
response into producible components and decreased use of demanding prompts were the main changes
made to RE/PMT during this study.
Results: Child and parent communication responses were measured before intervention and at 3
subsequent observation sessions conducted at 6-month intervals. After 6 months, children in the
intervention group produced significantly more communication acts than children that did not receive RE/
PMT. This effect was significant for both children with and without DS. However, this effect did not
maintain after treatment was discontinued. Furthermore, over time, children with DS showed relatively
slow growth in a number of communication variables, notably the number of different words produced,
regardless of whether they had participated in the RE/PMT intervention.
Discussion. The finding of significantly increased communication rates in response the modified version
of RE/PMT was encouraging. However, the lack of longitudinal effects indicates the need for further
research aimed at determining the necessary levels of intervention intensity and duration to achieve long
term growth.
This research was supported by USDE grant # H324CC990091 and NICHD P30-02528.
154
SYMPOSIUM 11
Effects of Early Augmented Language Intervention on the Communication Skills of Young
Children with and without Down Syndrome
Mary Ann Romski1,2, Rose A. Sevcik,2, Lauren B. Adamson,2,
Melissa Cheslock,1, Ashlyn Smith,2, Mike Barker,2, and Roger Bakeman,2
Depts. of Communication,1 and Psychology,2, PO Box 4000,
Georgia State University, Atlanta, Georgia 30302-4000,
Contact: [email protected]
Introduction: The ability of young children with significant developmental disabilities to acquire and use
language to control and to interact in their worlds is compromised significantly by their overwhelming lack
of speech production abilities. Early augmented language experiences that include speech generating
communication devices may facilitate communication development, prevent the emergence of challenging
behaviors, and facilitate social interactions with adults and peers. Little is known about the communication
development of toddlers with significant developmental delays with and without Down syndrome who
receive augmented language experience. In this presentation, we compare the effects of two parentimplemented augmented language interventions with a parent-implemented spoken language intervention
on the communication development of 18 children with Down syndrome and 42 children with other
developmental disabilities and significant delays in spoken language abilities.
Methods: The 60 toddlers ranged in age from 24 to 35 months (mean CA = 29.33 months) and evidenced
a significant expressive speech-language impairment operationally defined as an intentional communicator
but with less than 10 spoken words at the onset of the study. The majority of the children (80%) also
evidenced significant developmental delay defined as performance below the 1st percentile on the Mullen
Scales of Early Learning (Mullen, 1995). Participants were randomly assigned to one of three parentimplemented communication interventions: Spoken Communication Interaction (SCI), Augmented
Communication Input (ACI), or Augmented Communication Output (ACO). All three interventions were 12
weeks in length and focused on parent implementation of the intervention protocol first in the lab setting
and then at home. Children were assessed at pre- and post-intervention on a set of standardized and
experimental language and communication development measures. The children with Down syndrome
were distributed across the three intervention groups.
Results and Discussion: Young children with significant developmental disabilities learned to
communicate better with their parents in each of the three parent-implemented interventions than they did
at the start of the intervention. Both augmented language interventions, ACI and ACO, provided a way for
the children to communicate via visual-graphic symbols + digitized words after only 9 weeks of
intervention. Compared to the SCI intervention, ACO, and to a lesser extent ACI, significantly increased
the likelihood that children could produce spoken words for the target vocabulary after 9 weeks of parentimplemented intervention. Children with Down syndrome showed comparable symbol and word use to the
children without Down syndrome. Findings will be discussed with respect to the role augmented language
interventions can play in early language development of children with DS.
This research was supported by DC-03799 from the NIDCD.
155
S y m p o s i u m 12
S Y M P O S I U M
12
What Large-Scale, Administrative Databases Can Tell Us
about Down Syndrome
Chair: Robert M. Hodapp, PhD
Vanderbilt Kennedy Center for Research on Human
Development, Department of Special Education,
Peabody College, Vanderbilt University
156
SYMPOSIUM 12
What Large-Scale, Administrative Databases Can Tell Us about Down Syndrome
Chair: Robert M. Hodapp
Vanderbilt Kennedy Center for Research on Human Development
Department of Special Education, Peabody College, Vanderbilt University
Using State Administrative Databases to Study Down Syndrome
Richard C. Urbano
Vanderbilt Kennedy Center for Research on Human Development
Department of Pediatrics, Vanderbilt University Medical Center
The Demography of Births in Down Syndrome
Robert M. Hodapp
Vanderbilt Kennedy Center for Research on Human Development
Department of Special Education, Peabody College, Vanderbilt University
Down Syndrome and Adverse Birth Outcomes
S. Trent Rosenbloom
Vanderbilt Kennedy Center for Research on Human Development
Department of Biomedical Informatics
Propensity Scores: A Method for Matching on Multiple Variables in Down Syndrome
Jennifer U. Blackford
Vanderbilt Kennedy Center for Research on Human Development
Department of Psychiatry, Vanderbilt University Medical Center
Supported by NICHD grants P30HD 15052 and RO3HD 050468 (Hodapp & Urbano) and K22LM
08376 (Rosenbloom), as well as a Greenfield Discovery Grant from the Kennedy Center’s
Nicholas Hobbs Society (Hodapp).
157
SYMPOSIUM 12
Using State Administrative Databases to Study Down Syndrome
Richard C. Urbano
Vanderbilt Kennedy Center for Research on Human Development
Department of Pediatrics, Vanderbilt University Medical Center
Down syndrome is the most frequently occurring genetic syndrome, occurring in 1/800 to 1/1000 live
births. In Tennessee (5.8 million residents; 80,000 births per year), one would expect about 80-100 births
a year with a diagnosis of Down syndrome. Thus, statewide birth cohorts covering multiple years are
needed to obtain adequate samples.
State-level vital records provide a valuable resource for finding individuals with Down syndrome. For
conditions identified at birth, like Down syndrome, state birth vital statistics datasets are particularly useful.
Beyond identifying a population of such individuals, administrative records may be linked to create
comprehensive, multidimensional person-profiles. Person-profiles may contain information from multiple
sources for a single period (e.g., events surrounding birth or death) or information from multiple
observations over time (multiple hospitalizations of the same person). Person profiles may also be linked
to create family groups.
Method: In Tennessee, official, electronic Birth Records contain congenital anomalies identified at birth,
multiple individual identifiers of the child and parents, and comprehensive pregnancy, delivery, health and
demographic variables, for birth cohorts from 1990. Divorce records, also from 1990, were used to
examine the amount and correlates of divorce in families of children with Down syndrome.
Our Down syndrome research group has created several analysis datasets with person and family profiles.
Family divorce status was added to birth records by linking state divorce records to birth records to study
divorce in Down syndrome. Family groups were created by linking births to the same mother, allowing the
calculation of family level variables (e.g., # of boys, # of girls, boys/girls older/younger than the child with
Down syndrome). Each individual’s Hospital Discharge Records (originally one record per admission)
were linked together to create person-based hospitalization histories.
Results: From the congenital anomalies codes in the 1990-2002 Birth Records, we identified 550 children
with Down syndrome. In addition to the electronic birth records, the state maintains electronic records of
all inpatient and outpatient hospitalizations in Tennessee from 1996. Using individual identifiers in birth
and hospital discharge records, we were able to link birth and hospitalization data to identify an additional
368 births with an ICD-9 code of 758.0 (Down syndrome) in one or more hospital records. Given the rate
of Down syndrome, we would expect to find 1056 (1/1000) to 1320 (1/800) individuals. The 918 identified
individuals are 70% to 87% of the expected numbers. Additional studies linked multiple Hospital
Discharge Records of persons with Down syndrome; we are currently linking Birth Records to Hospital
Discharge Records and Birth & Hospital Discharge records to Vanderbilt electronic health records.
Conclusion: Large-scale administrative databases have historically been under-utilized in studies of
Down syndrome. Using such databases, we are beginning to examine a host of family, medical, and other
issues in this population.
158
SYMPOSIUM 12
The Demography of Births in Down Syndrome
Robert M. Hodapp
Vanderbilt Kennedy Center for Research on Human Development
Department of Special Education, Peabody College, Vanderbilt University
Most professionals know two facts about Down syndrome. First, Down syndrome occurs proportionally
more often to older mothers. Second, like most genetic disorders, Down syndrome equally affects births
to mothers in all races and educational levels.
In this talk, Dr. Hodapp uses Tennessee Birth Records to explore the implications of these two, somewhat
contradictory, facts. He explores the common finding that, compared to families of children with other
disorders, families of children with Down syndrome are more often White, more well-educated, and of
higher SES.
Method: This study utilizes linked Birth Records (1990-2002) and Hospital Discharge Records (19962003) to identify 918 newborns with Down syndrome in Tennessee. These infants were compared to
1,054,723 additional “Tennessee population” infants. Official Birth Records contain 140 pieces of
information, including the mother’s name, social security number, age, race, highest level of education,
and exact residence. Hospital Discharge Records were used to “track backward” those infants not found
in the official Birth Records (= 40% of all DS births). Our identification rates (0.087%) equaled from 70% to
87% of the 1/1000 to 1/800 incidence rate for Down syndrome.
Results: Across the population, fewer births occurred to mothers aged 35 to 39 (7.3%) and 40 and above
(1.4%); higher rates occurred for mothers below 20 (15.3%), 20 to 24 (30.3%), 25 to 29 (27.9%), and 30 to
34 (18.5%). Maternal age and education levels are also highly correlated (r = .48, p < .0001). Compared
to White mothers, African-American mothers gave birth at younger ages (X = 24.25 vs. 26.22 years; t =
143, p < .0001) and averaged fewer years of formal education (12.13 vs. 12.78, t = 141, p < .0001).
Combining these findings, it becomes clear why studies find that mothers of children with Down syndrome
are older, more educated, and more likely to be White. Of all Down syndrome births, proportionally more
occurred to mothers aged 35 to 39 (19.4% vs. 7.3% in the population) and aged 40 and above (12.2% vs.
1.4%). In contrast, fewer Down syndrome births occurred to women below 20 (9.6% vs. 15.3%) and from
20 to 24 (19.0% vs. 30.3%), x2= 1,043, p < .0001.
Given the correlation of maternal age and education, mothers of newborns with Down syndrome were
more educated. Over one-quarter (25.3%) had BA’s or beyond, compared to 18.4% of mothers in the
general population. Conversely, whereas 22.7% of population mothers had not finished high school, only
17.3% of mothers of newborns with Down syndrome were not high-school graduates, x2= 37.75, p <
.0001.
Racial disparities also occur in Down syndrome births. Births to White mothers comprised 76.5% of all TN
population births, 82.8% of all Down syndrome births. In contrast, African-American mothers accounted
for 21.9% of population births, 15.8% of Down syndrome births, x2= 20.63, p < .0001. Considering all
Down syndrome births in the African-American group, 41.3% were to mothers aged 24 and younger.
Conclusion: Beyond explaining the “noise” of family studies, population-based data help describe the
Down syndrome population for policy and intervention purposes.
159
SYMPOSIUM 12
Down Syndrome and Adverse Birth Outcomes
S. Trent Rosenbloom
Vanderbilt Kennedy Center for Research on Human Development
Department of Biomedical Informatics
Although infant mortality in the syndrome has definitely decreased over time, Down syndrome continues to
predispose newborns to a variety of adverse birth outcomes. In this study, we examined whether
newborns with Down syndrome have different rates of low birth weight, prematurity, small for gestational
age, and infant mortality than the general population of newborn infants, and evaluated whether
demographic and prenatal practice variables were associated with these adverse birth outcomes.
Method. This study employed a retrospective analysis of Tennessee Birth Records from 1990 through
2002, which includes all infants born in the state of Tennessee. Infants with Down syndrome were
identified using birth records and hospital discharge records (available from 1996 through 2003) of all
hospitalizations in the state. Birth records include information about the newborn (birth weight, estimated
gestational age, sex, birth order), mother (ethnicity, age, race, education), and maternal prenatal practices
(number and beginning month of prenatal doctor’s visits during pregnancy; weight gain during pregnancy;
whether the mother smokes). Outcomes included low birth weight; prematurity, small for gestational age,
and infant mortality.
Analyses compared the two groups; within Down syndrome, predictors were also identified of adverse
birth outcomes. For comparisons of newborns with Down syndrome versus those in the general
population, analyses involved both individual chi-square and t-tests. To determine independent predictors,
omnibus logistic regressions were performed using as outcomes low birth weight (<2500 grams),
prematurity (<37 weeks), small-for-gestational age (defined as the 10th percentile or less by gender), and
infant mortality (death within the first year of life).
Results. Compared to newborns in the general Tennessee population, newborns with Down syndrome
had greater risks for being born at low birth weights (2924 versus 3264 grams, OR = 2.75; CI: 2.35 –
3.21), prematurity (OR = 2.48; CI: 2.13 – 2.88), and small for gestational age (37.3 versus 38.5 weeks, OR
= 2.24; CI: 1.91 – 2.63). Infant mortality rates were 7.08 times (CI: 5.33 – 9.29) higher in the Down
syndrome group. Within Down syndrome, maternal weight gains of below 25 pounds, maternal smoking,
and 0-5 prenatal physician visits related to adverse birth outcomes. In the multivariate analysis, maternal
weight gain was associated with all four adverse birth outcomes; the number of prenatal visits during
pregnancy was associated with low birth weight, prematurity, and infant mortality; maternal smoking was
associated with low birth weights; and, maternal age was associated with low birth weight.
Conclusion. As one of the first studies examining the amounts and correlates of adverse birth outcomes
among newborns with Down syndrome, this study highlights the interplay of the effects of Down syndrome
and of maternal prenatal practices. Newborn infants with Down syndrome are at increased risks for
adverse birth outcomes. The presence of adverse birth outcomes among newborns with Down syndrome
seems related to the syndrome itself and to specific maternal practices during pregnancy.
160
SYMPOSIUM 12
Propensity Scores: A Method for Matching on Multiple Variables in Down Syndrome
Jennifer U. Blackford
Vanderbilt Kennedy Center for Research on Human Development
Department of Psychiatry, Vanderbilt University Medical Center
Families with a child with Down syndrome differ from the general population on a number of maternal
characteristics and demographic factors. In order to understand observed group differences beyond what
can be predicted by such demographic differences, researchers attempt to create appropriate matched
control groups. Even with large numbers of potential matches in a population sample, it can still be
challenging to directly match with categorical and/or continuous variables or control by including multiple
covariates. In this talk, Dr. Blackford introduces propensity scores based on multiple variables to select
control participants from a large database matched to a group with Down syndrome.
Method: Tennessee Birth (2001) and Hospital Discharge Records (2001-2003) were used to create a
large scale, administrative database including demographic variables, birth variables, and identification of
Down syndrome. Of the 83,039 births during the 2001 calendar year, 85 (0.102%) had Down syndrome.
Propensity Scores and Matching: Propensity scores are an efficient compromise between two extreme
methods of matching: selecting a small number of variables for matching and including too many
covariates in analyses. Propensity scores were used to reduce the information in multiple covariates into
a single number. Propensity scores were computed for each member of the birth cohort using a logistic
regression. Group membership (DS vs. non-DS) served as the dependent variable and predictor variables
included those within the Birth dataset related to risks of either having Down syndrome or for low
birthweight (maternal age, maternal education, number of prior births, plural birth, maternal weight gain,
maternal race (white/black), marital status, and child gender). For each individual in the DS group, an
individual in the non-DS group with a “close” propensity score was designated a match. A simple t-test of
the difference in birth weight between the DS and matched non-DS group was computed. Note that the
dependent variable birthweight was not used to create the propensity score.
Results: Using traditional, direct matching methods, only 45% of the DS births were successfully
matched. Using propensity scores, 91% (n=77) of the DS births were matched. In the unmatched
comparison of DS vs non-DS, mean birthweights were 2855 grams (SD=762) and 3244 grams (SD=627)
respectively. In the matched comparison, mean birthweights were 2825 (SD=89) and 3111 (SD=808),
respectively. While both comparisons reveal large, statistically significant differences, the effect size in the
unmatched comparison was .56, as opposed to .36 in the matched comparison.
Conclusion. 1) Even with relatively large datasets, propensity scores provide an efficient method for
creating matched control groups for studying health outcomes for children with Down syndrome. 2) Birth
weight differences between newborns with and without Down syndrome still exist even when using
propensity scores.
161
INDEX
A
D
Abbeduto, L ......................... 9, 12, 19, 20, 69, 151, 152
Adamson, LB ....................... 19, 152, 155
Adayev, T ............................. 10, 51, 54
Akai, CE .............................. 8, 22, 28
Albertini, G .......................... 10, 51, 54
Alessandri, M ...................... 11, 63
Alexander, ML ..................... 15, 104
Anderson, JA ....................... 12, 69
Antonarakis, SE .................. 10, 51, 53
Arancio, O ........................... 10, 51, 53
Dagnan, D ........................... 16, 117
Dalton, A .............................. 10, 51, 54
Danov, S .............................. 14, 93, 95
Davisson, MT ...................... 10, 51, 53
Day, J ................................... 13, 81
De Camilli, P ....................... 10, 51, 53
Della Rosa, A ...................... 11, 64
DeRamus, M ....................... 11, 56
Deutsch, CK ........................ 14, 88, 91
Devenny, DA ....................... 14, 16, 82, 83, 84, 85, 113
Di Paolo, G .......................... 10, 51, 53
Dommestrup, A ................... 18, 133
Doody, M ............................. 9, 36, 38
Dowjat, KW ......................... 10, 51, 54
Dube, WV ............................ 12, 14, 68, 88, 91
Dunn, ML ............................. 16, 109
Durocher, JS ....................... 11, 63
Dykens, EM ......................... 14, 17, 88, 89, 123
B
Bailey, Jr, D ......................... 12, 17, 71, 128
Bakeman, R ........................ 19, 152, 155
Baker, BL ............................. 15, 107
Ball, S .................................. 16, 114
Baranek, GT ........................ 11, 17, 56, 121
Barker, RM .......................... 17, 19, 120, 152, 155
Baron, A ............................... 12, 69
Barrio, J ............................... 14, 83, 86
Barua, M .............................. 10, 51, 54
Bates, MJ ............................ 12, 66
Becker, A ............................. 12, 67
Belser, RC ........................... 11, 13, 57, 79
Benson, PR ......................... 15, 99
Bialik, M ............................... 14, 88, 89
Biasini, FJ ............................ 12, 66
Blacher, J ............................. 15, 19, 107, 146, 147
Black, D ............................... 11, 64
Blackford, JU ....................... 19, 157, 161
Bodfish, JW ......................... 18, 94, 95, 97, 135
Boelter, E ............................. 18, 141
Borel, C ................................ 10, 51, 53
Borkowski, JG ..................... 8, 17, 21, 22, 24, 27, 124
Boyd, BA ............................. 11, 17, 56, 121
Brady, NC ............................ 10, 11, 13, 19, 45, 46, 58, 78, 152, 154
Bredin-Oja, SL ..................... 19, 152, 154
Brown, WT ........................... 15, 100
Burke Lefever, J .................. 8, 22, 23
Burns, L ............................... 13, 81
C
Carolyn Graff, J ................... 9, 36, 39
Carothers, S ........................ 8, 22, 25
Carta, JJ .............................. 8, 22, 24
Carter, I ................................ 11, 59
Carter, S .............................. 14, 88, 89
Chen, C ............................... 14, 93, 94, 95
Cheslock, M ........................ 19, 152, 155
Chudley, A ........................... 15, 100
Cimasoni, L ......................... 10, 51, 53
Clary, J ................................. 94, 95
Cohen, IL ............................. 15, 100
Cohn, ES ............................. 16, 109
Cole, A ................................. 11, 60
Corrice, AM .......................... 17, 122
Coster, J .............................. 11, 61
Coster, WJ ........................... 16, 109
Costigan, FA ........................ 18, 135
Crittenden, K ....................... 41, 42
Crnic, K ................................ 18, 136
Crowson, SB ....................... 12, 66
162
E
Emerson, E ......................... 15, 16, 108, 110
Esbensen, AJ ...................... 11, 62
F
Faria, A ................................ 17, 130
Farris, JR ............................. 17, 124
Fenning, R ........................... 15, 107
Fey, ME ............................... 19, 152, 154
Fidler, DJ ............................. 11, 12, 18, 60, 70, 137
Finestack, LH ...................... 19, 152, 154
Fleming, KK ......................... 11, 19, 58, 152, 154
Flory, M ................................ 9, 41, 42
Floyd, FJ .............................. 13, 18, 74, 77, 140
Frere, SG ............................. 10, 51, 53
G
Gaines Lanzi, R ................... 8, 22, 23, 25
Gardiner, K .......................... 10, 51, 53
Gardner, JM ......................... 11, 13, 17, 57, 79, 129
Gay, E .................................. 17, 121
Giles, ND ............................. 12, 69
Gilles, E ............................... 97
Giovedi, S ............................ 10, 51, 53
Glidden, LM ......................... 9, 11, 13, 17, 40, 41, 61, 73, 122
Gong, C ............................... 10, 51, 54
Gonzalez, M ........................ 15, 100
Gonzalez, V ......................... 11, 63
Gordon, A ............................ 17, 129
Greenberg, JS ..................... 11, 62
Greenslade, K ..................... 12, 67
Grey, I .................................. 9, 36, 38
Gross, AM ............................ 17
Grundke-Igbal, I .................. 11, 51, 54
Gu, H ................................... 16, 113
Gu, W .................................. 10, 51, 52
Guest, KC ............................ 8, 22, 28
Gulsrud, A ............................ 10, 46, 48
H
Hagopian, LP ...................... 18, 141
Hancock, TB ........................ 10, 47
Har, K ................................... 17, 125
Hardcastle, B ....................... 16, 115
Harin, A ................................ 17, 129
Harper, V ............................. 94, 95
Harrison, B .......................... 11, 64
Hartley, SL ........................... 17, 126
Harvey, MT .......................... 12, 67
Hastings, R .......................... 9, 13, 17, 19, 36, 38, 81, 131, 146, 148
Hatton, C ............................. 15, 16, 108, 110
Hatton, DD ........................... 15, 102, 103
Hauser-Cram, P .................. 16, 116
Hayes, A .............................. 12, 65
Head, LS .............................. 17, 127
Henderson, LM .................... 15, 101
Hepburn, SL ........................ 11, 12, 18, 60, 65, 70, 137
Herbert, J ............................. 16, 114
Hoch, J ................................ 14, 93, 96
Hodapp, RM ........................ 9, 19, 36, 37, 156, 157, 159
Holden, JJA ......................... 15, 100
Holland, AJ .......................... 14, 16, 82, 83, 114
Hopkins, IM ......................... 12, 66
Howard, KS ......................... 8, 22, 24
Hughes, KP ......................... 8, 22, 24
Hurlbut, D ............................ 12, 68
Huws, J ................................ 18, 138
Hwang, Y ............................. 10, 51, 54
I
Imaki, H ............................... 10, 51, 54
Iqbal, K ................................ 11, 51, 54
J
Jahoda, A ............................ 13, 16, 76, 117
Jahromi, LB ......................... 10, 46, 48
James, A .............................. 17, 128
Jenkins, EC ......................... 9, 15, 16, 30, 32, 100, 113
Jennett, HK ......................... 18, 141
Jobe, BM ............................. 11, 13, 17, 61, 73, 122
Johnson, R .......................... 14, 17, 88, 89, 123
Johnstone, EC ..................... 16, 111
K
Kaczmarski, W .................... 10, 51, 54
Kaiser, AP ............................ 9, 10, 35, 36, 46, 47
Kaiser, MY ........................... 11, 63, 130
Karmel, BZ .......................... 11, 13, 17, 57, 79, 129
Kasari, C .............................. 10, 17, 46, 48, 125
Keltner, BR .......................... 8, 22, 25, 28
Kennedy, CH ....................... 12, 67
Kennedy, W ......................... 97
Kenworthy, L ........................ 11, 64
Kepe, V ................................ 14, 83, 86
Kersh, J ............................... 16, 116
Kim, S-Y .............................. 15, 100
Kittler, P ............................... 14, 17, 83, 84, 85, 129
Kledaras, JB ........................ 12, 68
Klerman, LV ......................... 8, 22, 28
Kolnik, S .............................. 17, 130
Kon, N .................................. 10, 51, 52|
Kossler, R ............................ 14, 17, 88, 89, 123
Kover, ST ............................. 12, 69
Kovshoff, H .......................... 17, 131
Krinsky-McHale, SJ ............ 9, 14, 30, 33, 83, 84, 85
Kuchna, I ............................. 10, 51, 54
Kupersmidt, JB .................... 13, 74, 77
L
Landesman Ramey, S ........ 8, 22, 25
Lee McIntyre, L ................... 18, 133
Lewis, P ............................... 12, 69
Lewis, S ............................... 15, 100
Li, M ..................................... 10, 51, 52
Liang, Z ................................ 11, 51, 54
Lieving, GA .......................... 18, 141
Lin, CS ................................. 10, 51, 52
Liu, F .................................... 11, 51, 54
Liu, X .................................... 15, 100
Lloyd, T ................................ 13, 19, 75, 146, 148
Lott, I .................................... 6, 14
Lubke, G .............................. 8, 22, 26
M
MacLean, Jr., WE ............... 12, 17, 65, 126
MacWhorter, KJ .................. 18, 141
Magruder, H ......................... 18, 132
Mahoney, G ......................... 15, 106
Mailick Seltzer, M ................ 9, 11, 20, 62
Malesa, EE .......................... 12, 72
Malow, BA ............................ 15, 101
Mankowski, JB .................... 12, 71
Marzec, ML .......................... 15, 101
Mayeux, RP ......................... 8, 30, 31
Mazzocco, MMM ................. 18, 134
McCann, J ........................... 16, 115
McGrew, SG ........................ 15, 101
McIlvane, WJ ...................... 12, 14, 68, 88, 91
McMahon, CR ..................... 12, 72
Mehta, PD ............................ 8, 10, 30, 31, 51, 54
Moffat, VJ ............................ 16, 111
Morris, RD ........................... 18, 144
Most, D ................................ 11, 60
Mulligan, J ........................... 13, 81
Murakami, N ........................ 11, 51, 54
Murphy, MM ......................... 18, 134
N
Nash, S ................................ 13,
Natcher, AL .......................... 13,
Nazarloo, H ......................... 14,
Neece, C .............................. 19,
Nelson, LD ........................... 14,
Neudorfder, NJ .................... 18,
Newell, KM .......................... 18,
Ngai, I .................................. 13,
Ni, SA .................................. 16,
Nicholson, JS ...................... 17,
Noone, S .............................. 13,
Nowicki, K ............................ 10,
75, 80
73
88, 89
146, 147
83, 86
133
135
74
113
124
81
51, 54
163
O
O’Neill, S ............................. 9, 36, 38
Olsson, MB .......................... 19, 145, 146, 150
Orsmond, GS ...................... 9, 16, 35, 36, 109
P
Pablo Juarez, A ................... 12, 67
Pan, H .................................. 16, 116
Pang, D ................................ 8, 16, 30, 31, 113
Pantino, E ............................ 14, 17, 88, 89, 123
Parker, D ............................. 94, 95
Parsa, RA ............................ 15, 104
Patel, BP ............................. 8, 30, 31
Pearson, RK ........................ 12, 66
Pedersen y Arbona, A ......... 18, 136
Pennington, B ...................... 12, 70
Perales, F ............................ 15, 106
Pert, C ................................. 16, 117
Petalas, MA ......................... 13, 75
Phan, HTT ........................... 17, 129
Philofsky, A .......................... 11, 12, 18, 60, 70, 137
Piazza, V ............................. 18, 140
Pitcairn, TK .......................... 18, 142
Poe, MD ............................... 11, 56
Pollina, EA ........................... 10, 51, 53
Pownall, J ............................ 13, 76
Tally, B ................................. 12, 67
Tarvainen, M ........................ 96
Thompson, T ....................... 14, 87, 88, 90
Timmins, C .......................... 16, 115
Tobin, EL ............................. 18, 141
Toogood, S .......................... 13, 80
Toole, LM ............................. 18, 141
Totsika, V ............................. 13, 80
Trower, P ............................. 16, 117
Tycko, B ............................... 9, 10, 30, 32, 50, 51, 52
Urbano, RC ......................... 9, 19, 36, 37, 157, 158
Urv, TK ................................. 9, 30, 33, 34
V
103, 128
137
49, 152, 155
123
S
Salas, M ............................... 10, 51, 52
Sallows, G ........................... 14, 88, 90
Schatz, JN ........................... 8, 22, 24, 27
Schmidt, C ........................... 10, 51, 53
Schroeder, S ....................... 12, 69
Schupf, N ............................. 8, 9, 10, 16, 30, 31, 32, 34, 50, 51, 113
Selkirk, M ............................. 16, 117
Sevcik, RA ........................... 16, 18, 19, 120, 144, 152, 155
Shanahan, M ....................... 15, 103
Sherman, S ......................... 7, 9
Shin, JY ............................... 9, 40, 41, 42
Shinde, S ............................. 95
Siddarth, P ........................... 14, 83, 86
Sideris, J .............................. 15, 102
Silverman, WP .................... 8, 9, 10, 11, 14, 16, 29, 30, 31, 32, 33,
............................................. 34, 51, 52, 54, 83, 85, 113
164
T
U
R
Rabe, A ................................ 11, 51, 54
Raitano Lee, N .................... 12, 70
Ramakrishna, N .................. 11, 51, 54
Ramey, C ............................. 8, 21, 22
Reeves, RH ......................... 6, 20
Reilly, D ............................... 18, 138
Remington, B ...................... 17, 131
Richmond, EK ..................... 12, 69
Roberts, JE .......................... 12, 15, 17, 71,
Rogers, SJ ........................... 12, 18, 65, 70,
Rojas, C ............................... 11, 63
Romski, MA ......................... 10, 19, 45, 46,
Roof, E ................................. 14, 17, 88, 89,
Rosenbloom, ST ................. 19, 157, 160
Rovito-Gomez, T ................. 15, 100
Ryan, R ................................ 9, 30, 34
Small, G ............................... 14, 83, 86
Smith, AL ............................. 10, 19, 46, 49, 152, 155
Smith, LE ............................. 8, 22, 26, 28
Snead, K .............................. 13, 77
Spillane, A ............................ 18, 139
Stenfert-Kroese, B .............. 16, 117
Sterling, A ............................ 13, 78
Stone, WL ............................ 12, 15, 72, 101
Stoneman, Z ........................ 19, 146, 149
Strickland, S ........................ 18, 140
Sudhalter, V ......................... 11, 13, 57, 79
Sullivan, C ........................... 12, 67
Sullivan, K ........................... 15, 102
Symons, FJ ......................... 14, 92, 93, 94, 95, 96, 97
Valdovinos, MG ................... 15, 104
Valenti, SS ........................... 9, 41, 42
Vaughan, F .......................... 18, 138
Velinov, MT .......................... 16, 113
Viet Nhan, N ........................ 9, 41, 42
Vijay, P ................................. 10, 47
Voronov, SV ......................... 10, 51, 53
W
Wallace, G ........................... 11, 64
Wang, L ............................... 15, 101
Wang, P ............................... 10, 41, 44
Warren, SF .......................... 8, 13, 19, 22, 26, 78, 151, 152, 153,
............................................. 154
Watson, LR .......................... 11, 17, 56, 121
Weber, A .............................. 12, 69
Wegiel, J .............................. 10, 51, 54
Wendelschafer-Crabb, G .... 97
Wenk, MR ............................ 10, 51, 53
Wheeler, A ........................... 15, 102
Wilkinson, KM ..................... 14, 88, 91
Williams, KR ........................ 18, 142
Williams, L ........................... 13, 81
Willis, DS ............................. 18, 142
Wilson .................................. 13, 76
Wise, JC .............................. 18, 144
Wishart, JG ......................... 7, 15, 16, 18, 111, 115, 142
Wong, CS ............................ 18, 143
Wood, S ............................... 16, 115
X
Xing, L ................................. 10, 51, 52
Y
Yale Kaiser, M ..................... 17
Ye, L ..................................... 16, 113
Yoder, PJ ............................. 12, 19, 72, 152, 153
Young Ryu, S ...................... 10, 41, 43
Z
Zigman, WB ........................ 8, 9, 10, 16, 30, 31, 32, 33, 34, 51, 52, 113
165
Notes
Notes