Effect of treatment for bilingual individuals with aphasia: A systematic review of the evidence Yasmeen Faroqi-Shaha, Tobi Frymarkb, Robert Mullenb, Beverly Wangb a b University of Maryland, College Park, MD, USA National Center for Evidence-based Practice in Communication Disorders, American SpeechLanguage Hearing Association, Rockville, MD, USA *Corresponding Author: Tobi Frymark National Center for Evidence-based Practice in Communication Disorders American Speech-Language Hearing Association 2200 Research Blvd, Rockville, MD 20850 301-296-8742 (o), 301-296-8588 (f) [email protected] This is a preprint text of an article accepted for publication in the Journal of Neurolinguistics. Please cite this article in press as: Faroqi-Shah, Y. et al., Effect of treatment for bilingual individuals with aphasia: A systematic review of the evidence, Journal of Neurolinguistics (2010), doi:10.1016/j.jneuroling.2010.01.002 Abstract Language proficiency in bilingualism, and hence bilingual aphasia, is a multifaceted phenomenon: influenced by variables such as age of onset, literacy, usage patterns, and emotional valence. Although the majority of the world and growing US population is bilingual, relatively little is known about the best practices for language therapy in bilingual aphasia. This systematic review was undertaken to examine three crucial questions faced by speech-language pathologists during clinical decision making: outcomes when language therapy is provided in the secondary (less-dominant) language (L2), extent of cross-language transfer (CLT) and variables that influence CLT, and outcomes when language therapy is mediated by a language broker. Data from 14 studies (N=45 aphasic individuals) indicate that treatment in L2 leads to positive outcomes (akin to L1 treatment); CLT was found to occur in most studies, especially when L1 was the language of treatment. Although limited by the methodological quality of included studies, this systematic review shows positive findings for unilingual aphasia treatment and CLT. Implications for clinical practice, models of language representation in bilinguals, and future research directions are discussed. Key Words: Aphasia; Bilingualism; Cross-Language Transfer; Multilingualism; SpeechLanguage Pathologist 1. Introduction More than half the world (and a rapidly growing US demographic) is bilingual1. Hence the occurrence of bilingual aphasia is more common than what can be gleaned from the literature. Speech-language pathologists (SLPs) are increasingly likely to provide services to bilingual aphasic clients (Ansaldo, Marchotte, Scherer, & Raboyeau, 2008; Centeno, 2009; Paradis, 2001). Given that the overarching goal of language treatment for individuals with aphasia is to achieve the maximum possible level of life participation, the goal of language treatment in the bilingual client becomes the improvement of communication in both languages. Providing language treatment to bilingual clients may pose challenges that are less evident when providing treatment to monolingual clients with aphasia. The obvious logistical challenges include access to bilingual assessment and treatment materials and availability of bilingual SLPs, or two SLPs speaking the relevant languages. There is also an important and largely unresolved conceptual challenge in the treatment of bilingual aphasia — whether to focus on a single language or include both languages in treatment. Experts who recommend bilingual therapy point out that inclusion of both languages ensures that the aphasic person is able to utilize all possible communicative strategies available to him/her (akin to using gesture or writing to aid verbal communication) (Ansaldo et al., 2008; Centeno, 2005; Kohnert, 2004). It is also argued that the bilingual speaking environment is the most natural for some bilinguals; thus, bilingual therapy is the best choice. However, some authors point out certain caveats and suggest that bilingual therapy can lead to increased code mixing-code switching, or could suppress 1 We use the term bilingual as used by Grosjean (1994) to refer to all individuals who use two or more languages or dialects in their daily communicative environment, irrespective of the context of use. recovery of one language (Hemphill, 1976; Lebrun, 1988). In fact, there are reports of bilingual treatments leading to improvement of only one language (Paradis, 1993). From a neurolinguistic perspective, bilinguals possess an intermixed lexical and morphosyntactic organization (Golesteni et al., 2006; Gollan, Montoya, Fennema-Notestine, & Morris, 2005; Kroll & Stewart, 1994). The intermixed neurolinguistic organization is not only used to make the case for bilingual therapy, but can also be used to argue that therapy in a single language (henceforth unilingual therapy) will automatically transfer to the untrained language because of stimulation of shared neural networks (Kohnert, 2009; Watamori & Sasanuma, 1978). Unilingual therapy is also recommended for individuals who experience pathological code mixing-code switching or who live in a primarily monolingual environment (Abutelabi & Green, 2008; Ansaldo, Ghazi Saidi, & Ruiz, 2009). However, this prediction of cross-language transfer (CLT) with unilingual therapy has not been consistently borne out (e.g., Edmonds & Kiran, 2006; Faroqi & Chengappa, 1996; Filiputti, Tavano, Vorano, Luca, & Fabbro, 2002). Discussions of variables that influence success of CLT have questioned whether the first (L1) and second (L2) languages are equipotent in their prospects for language gains. One proposal is that language proficiency may interact with CLT potential such that low proficiency bilinguals are more likely than high proficiency bilinguals to experience CLT after unilingual therapy in L2 (Edmonds & Kiran, 2006). This is because the L2 of low proficiency bilinguals depends to a greater extent on borrowings from L1; while the L2 of high proficiency bilinguals is relatively independent of L1 (Jared & Kroll, 2001). However, CLT effects with L2 therapy are not always reported. The foregoing discussion raises several pertinent questions that are unresolved. Namely, do bilingual aphasic clients benefit from treatment provided in their L2? Does unilingual therapy result in CLT? Do L1 and L2 differ in CLT potential? And do any factors (demographic, linguistic, aphasia-related, or otherwise) help predict success with L2 therapy and CLT? A cursory Medline search using the terms bilingualism and aphasia reveals 89 citations; the majority of which characterize the nature and recovery pattern of bilingual aphasia (Fabbro, 2001; Green, 2005; Levy, Goral, & Obler, 1999; Lorenzen & Murray, 2008; Goral, Levy & Obler, 2002; Obler & Mahecha, 1991) with only a small number focused specifically on the impact of treatment. This superficial look at the literature does not provide straightforward answers to the previously raised questions. Therefore a more comprehensive and meticulous examination of the literature is warranted2. This paper describes the findings of an evidence-based systematic review (EBSR) conducted by the American Speech-Language-Hearing Association’s (ASHA’s) National Center for Evidence-based Practice in Communication Disorders. The primary aim of this review is to synthesize and analyze the existing data on aphasia treatment for bilingual individuals. Knowledge of the current evidence is likely to assist SLPs in therapeutic decision making. In addition, it is hoped that this review will serve to highlight the empirical strength of the current evidence (or lack thereof) and identify unresolved questions in need of further research. An essential first step in initiating a systematic review of the literature was to formulate the questions for data extraction. In constructing the clinical questions, it was decided that the impact of L1 therapy on L1 outcomes in bilingual individuals was not a crucial issue because this is analogous to examining the efficacy of aphasia therapy in the native language of monolingual 2 It should be noted that a review article by Kohnert (2009) addressing CLT was published after the completion of the present study. The authors were unaware of this article prior to the completion of the present review and variations in clinical questions addressed, number of databases searched and study inclusion parameters led to a minimal number of overlapping studies (5) reported. clients. And there is ample evidence of the success of aphasia therapy (Beeson & Robey, 2006; Holland, Fromm, DeRuyter, & Stein, 1996; Robey & Schultz, 1998). For this reason, we decided to focus on the effect of L2 therapy. Our second focus was to examine the occurrence of CLT in both directions (L1 to L2 and L2 to L1) and a third was to determine the effect of therapy that was mediated by a language broker when the therapist and client spoke different languages. Given that receptive and expressive language abilities can be relatively independent and treatment does not always generalize across both modalities, we decided to examine treatment effects on expressive and receptive language in separate analyses. Finally, we synthesized pertinent variables such as age of participant, age of L2 acquisition, pre-morbid proficiency in each language, language of the environment, aphasia characteristics, and time post onset to determine factors that might impact outcomes. This resulted in the following eight questions in three focus areas: Focus A: Language therapy in the secondary language (L2) 1. What is the effect of treatment provided by an SLP in L2 on the receptive language skills in the treated language (L2) for bilingual clients with neurologically-induced aphasia? 2. What is the effect of treatment provided by an SLP in L2 on the expressive language skills in the treated language (L2) for bilingual clients with neurologically-induced aphasia? Focus B: Cross-language transfer (CLT) of therapy outcomes 3. What is the effect of treatment provided by an SLP in L2 on the receptive language skills in the untreated language (L1) for bilingual clients with neurologically-induced aphasia? 4. What is the effect of treatment provided by an SLP in L1 on the receptive language skills in the untreated language (L2) for bilingual clients with neurologically-induced aphasia? 5. What is the effect of treatment provided by an SLP in L2 on the expressive language skills in the untreated language (L1) for bilingual clients with neurologically-induced aphasia? 6. What is the effect of treatment provided by an SLP in L1 on the expressive language skills in the untreated language (L2) for bilingual clients with neurologically-induced aphasia? Focus C: Therapy outcomes with a language broker 7. What is the effect of services provided by a language broker in L1 on the receptive language skills in the untreated language (L2) for bilingual clients with neurologically-induced aphasia? 8. What is the effect of services provided by a language broker in L1 on the expressive language skills in the untreated language (L2) for bilingual clients with neurologicallyinduced aphasia? 2. Method 2.1. Literature search A literature search was conducted during July and August 2009. Research studies were identified from 29 electronic databases using keywords pertaining to bilingualism or multilingualism and aphasia (see A.1 for a complete list of databases and Supplementary material for the expanded search terms). Inclusionary criteria that were used to determine eligibility were: research studies published in peer-reviewed journals from 1980 to August 2009 with original data pertaining to the EBSR question(s), publications in English language (due to ease of access and limited translation resources), and studies that included bilingual adults (ages 18 years or older) with neurologically-induced aphasia and described outcomes of language intervention. As mentioned earlier, bilingual individuals included all individuals who spoke two or more languages in their daily life, irrespective of manner and age of acquisition (Grosjean, 1992; Grosjean, 1994). Interventions included any SLP treatment conducted in primary (L1) or secondary (L2) language targeting receptive and/or expressive language skills. Exclusion criteria were studies that described individuals with cognitive deficits, studies that included participants with heterogeneous etiologies (unless data could be separated), and interventions that were pharmacological, or utilized augmentative and alternative communication. Two authors (RM and TF) independently reviewed all citations for relevance based on the predetermined inclusion criteria. References from all full-text articles and narrative reviews were also hand-searched and when necessary, the study authors were contacted to obtain original data or studies. Inter rater reliability between the two authors for study inclusion was determined using the kappa statistic (Cohen, 1960). Study eligibility agreement between RM and TF was good, K = .852. Disagreements were discussed and resolved by consensus. A third author (YF-S) reviewed the full list of accepted and rejected bibliographies for completeness prior to final inclusion/exclusion. Figure 1 schematizes the literature search. Of the 174 citations reviewed, 36 were identified for preliminary inclusion. After obtaining the full text of these articles, more than half (64%; 23/36) were further eliminated. One study preliminarily accepted (Fabbro, Deluca, & Vorano, 1996) could not be obtained despite attempted correspondence with authors. An additional treatment study (Filiputti, et al., 2002) could not be included in our data analysis because it only provided overall language measures (for morphology, syntax, etc.) that were derived by combining expressive and receptive scores. Therefore, the data could not be evaluated for a specific clinical question(s). A total of 161 citations were excluded from the review; the majority of which did not provide an intervention (50%, 81/161). Other reasons for exclusion were as follows: a) was not a study or systematic review (23%, 37/161), b) not age or population under review (6%, 10/161), c) did not provide original data or separate data from mixed populations or treatments (6%, 10/161), d) was not published in a peer-reviewed journal (7%, 12/161), e) did not target a question (6%, 10/161) or f) could not obtain full-text (1%, 1/161); leaving a total of 13 citations. One citation (Maragnolo, Rizzi, Peran, Piras, & Sabatini, 2009) provided data from two distinct studies resulting in 14 studies for review. The list of excluded articles with reasons for exclusion is provided as Supplementary material (see S2). ------------Insert Figure 1 about here---------Data extraction and coding Methodological quality of included studies was independently appraised by RM and TF on six indicators identified by ASHA’s levels of evidence scheme (ASHA, 2007). To minimize bias, studies were evaluated on whether or not they provided an adequate description of study protocol, whether assessors of outcomes were blinded to language of intervention, use of adequate sampling/allocation procedures, evidence of treatment fidelity, report of significance (p values) and report of precision (effect size and confidence intervals). Studies were not evaluated on ASHA’s seventh quality indicator, use of intention to treat, as no controlled trials were found in which this analyses was applicable. A description of quality indicators and corresponding quality markers are provided as Supplementary material (see S.3). Level of agreement between reviewers on study quality was good (K = 0.61 - 0.80; Landis & Koch, 1977). Each study was examined for the question(s) which it addressed and relevant pre- and post-therapy data were extracted. We computed statistical significance for the pre and post-treatment scores using the McNemar’s change test (p<0.05, Seigel & Castellan, 1988)3. There were two primary reasons for performing the statistical computations. Some studies failed to report any statistical measure (e.g., Faroqi & Chengappa, 1996; Gil & Goral, 2004; Khamis, Verkent-Olenik, & Gil, 1996). A few other studies reported parametric statistical tests whose assumptions of normality and independence were not met by the study design and data. McNemar’s change test is a nonparametric test for paired nominal measures such as accuracy data, and has been used by several aphasiologists to compute statistical significance of treatment-induced changes in behavioral scores (Faroqi-Shah, 2008; Rochon, Laird, Bose, & Scofield, 2005). The use of a consistent statistical measure makes comparisons of statistical significance across studies more valid. In addition, one author (TF) extracted several key elements from each study to determine the influence, if any, the participant’s linguistic, demographic and treatment characteristics have on language outcomes. Variables included age, gender, level of education, medical diagnosis, aphasia characteristics, time post onset, type, frequency, intensity and duration of treatment, person administering intervention, L1 and L2 language, acquisition of secondary language, level of pre-morbid proficiency in each language, and cognate relationships. Studies varied considerably in the types of treatment administered and the extent to which the treatment protocol was described. Broad categories of treatment types were coded on the basis of the focus of the intervention. Categories included auditory-comprehension, word-semantic, wordphonology, sentence-syntax, and non-specific. 2.3. Operational definitions 3 The following on-line calculator was used: http://www.graphpad.com/quickcalcs/McNemar1.cfm Unilingual treatment was operationally defined as the use of a single language at any given treatment phase. Studies that used alternating treatments with a separate language for each treatment phase were included. Studies that utilized both languages within a treatment phase or used translation between languages were excluded. We operationally defined L1 as the first acquired language as this was the criterion used by almost all the studies. It must be noted that in some studies the individual with aphasia pre-morbidly used the L1 with a lower frequency or proficiency than L2. Level of L1 and L2 proficiency was defined as high or low based on author’s report (i.e. pre-morbid reading, writing and conversational levels). L2 was operationally defined as the later acquired language, and it must be noted that this definition includes treatment provided in a third or fourth acquired language. Early and late bilingualism was defined as L2 acquisition before or after five years of age respectively. Receptive language was defined as any task that tapped input modalities (auditory or visual) such as auditory discrimination, lexical decision, sentence comprehension, and picture identification. Expressive language was defined as tasks that included verbal output such as picture naming, sentence production, synonym generation, repetition, and picture description. Reading and writing tasks were excluded as these involve multiple modalities. A language broker was operationally defined as a person who lacked professional training (as an SLP or related profession) and served to mediate between an SLP and bilingual client by interpreting (oral language) and translating (written language). Acute and chronic aphasia were defined as within and after one year post-onset of the brain damage respectively. Any treatment with a primary focus on improving auditory comprehension was coded as auditory-comprehension. Treatments that focused on word retrieval were coded as word-semantic or word-phonological based on whether semantic attributes or phonemic cueing strategies were primarily used. The term sentence-syntax was used for treatments that focused on sentence production, either with single sentences or discourse. Studies in which the treatment description lacked sufficient detail for categorization were coded as non-specific. 3. Results 3.1. Overview Table 1 summarizes the questions addressed, quality markers, study design and number of participants for each of the studies included in this review; all of which were in the exploratory stage of research (ASHA, 2007; See S3). The data included covers a total of 45 bilingual aphasic participants, with the majority of participants (N=30) contributed by a single group study (Junque, Vendrell, Vendrell-Brucet, & Tobena, 1989). Eighty-six percent of the studies (12/14) contributed data to more than one question and 79% (11/14) provided data across more than one focus area. Of the 14 included studies, 12 addressed one or more questions in Focus area A (language therapy in the secondary language) and 13 addressed one or more questions in Focus area B (CLT of therapy outcomes). No study investigated outcomes with language brokers (questions 7 & 8) and hence questions under Focus area C could not be evaluated. Our systematic search of the literature found few studies (N = 6) pertaining to the effects of SLP treatment on receptive language skills (questions 1,3,4). All included studies addressed the effects of SLP treatment on expressive language skills (questions 2,5,6); the majority of which (79%; 11/14) investigated the direct effect of SLP treatment in L2 on the expressive language skills of the treated language (question 2). Eleven studies also examined cross linguistic gains on expressive language skills as a result of SLP treatment provided in L2 (question 5) and five studies examined CLT of treatment provided in L1 (question 6). ----------------Insert Table 1 here--------------- Appraisal of methodological quality revealed that most (13/14) described individual participant data as part of a case study or single subject design and had adequate description of the study protocol. However, studies lacked in other quality markers such as random sampling, blinding of assessors, and evaluation of treatment fidelity. Therefore the reader may note that the interpretive strength of this review is somewhat limited by the quality of the component studies (Dollaghan, 2007). In the following sections, the findings are presented under each focus area. Data on demographic and linguistic variables is described last. Unless otherwise noted, reports of statistical significance refer to this authors’ computation of significance using McNemar’s change test (p<0.05). 3.2. Focus A: Language therapy in the secondary language (L2) Five case studies investigated Question 1, the effect of unilingual treatment in L2 on receptive language skills in L2 (Abutelabi, Rosa, Tettamanti, Green, & Cappa, 2009; Faroqi & Chengappa, 1996; Gil & Goral, 2004; Kamis, Venkert-Olenik, & Gil, 1996; Miertsch, Meisel, & Isel, 2009). These are listed in Table 2a. Although none of the studies reported effect size or probability data, the participants in all five studies exhibited significantly increased performance in L2 receptive language skills after SLP treatment was provided in L2 (McNemar’s change test, p<0.05). Three of the studies described acute aphasic patients with primarily receptive difficulties and the primary aim of therapy was to improve auditory comprehension (Abutelabi, et al., 2009; Gil & Goral, 2004; Kamis et al., 1996). One study with a chronic patient also aimed at improving auditory comprehension in addition to lexical retrieval (Miertsch, et al., 2009). The Faroqi & Chengappa study (1996) differed from the above four studies in several aspects: the patient had primarily expressive difficulties with relatively spared word level comprehension (diagnosis of Broca’s aphasia as compared to Wernicke’s aphasia for the other four studies). The therapy focused on syntactically complex sentences and used a combination of receptive and expressive tasks. It should be noted that studies differed considerably in the extensiveness of language scores provided, and some studies listed only those pre- post- scores that differed significantly (e.g., Miertsch et al., 2009), while other studies provided a more complete listing of scores (e.g., Abutelabi et al., 2009). Across studies, commercially available instruments such as the Bilingual Aphasia Test (BAT; Paradis, 1987), Israeli Lowenstein Aphasia Test (ILAT; Schechter, 1965) and/or custom-made measures were used. This confounds the interpretation of outcome comparisons across studies. These caveats are addressed further in the discussion section. ---------------Insert Tables 2a and 2b about here--------------As shown in Table 2b, 12 studies contributed data relevant to Question 2, the effect of L2 treatment on L2 expressive language (Abutelabi et al., 2009; Edmonds & Kiran, 2006; Faroqi & Chengappa, 1996; Gil & Goral, 2004; Goral, Levy & Kast, 2009; Khamis et al., 1996; Laganaro, Di Pietro & Schnider, 2003; Marangolo et al., 2009 (study 1); Marangolo et al., 2009 (study 2); Meinzer, Obleser, Flaisch, Eulitz, & Rockstroh, 2007; Miertsch et al., 2009; Penn & Beecham, 1992). Outcome measures included the Aechen Aphasia Test (AAT; Huber, Poeck, & Williams, 1984), BAT, ILAT; Boston Naming Test (BNT; Kaplan, Goodglass & Weintraub, 1983), Snodgrass Naming Battery (SNB; Snodgrass & Vanderwart, 1980) and informal assessment measures. Eight of these studies reported data from acute aphasic participants, seven of which focused on word retrieval, and found statistically significant changes in word retrieval scores (McNemar’s change test, p<0.05) (Abutelabi et al., 2009; Edmonds & Kiran, 2006; Gil & Goral, 2004; Khamis et al., 1996; Lagarno et al., 2003; Marangolo et al., 2009 study 1 and 2). One case study of an acutely aphasic participant focused on discourse strategies and reported qualitative data and so statistical comparisons could not be made (Penn & Beecham, 1992). Two studies of chronic participants focused on word retrieval treatments and found statistically significant improvements (Meinzer et al., 2007; Miertsch et al., 2009). The remaining two chronic studies examined the efficacy of morphosyntactic and syntactic treatments respectively (Faroqi & Chengappa, 1996; Goral et al., 2009). The morphosyntactic treatment changed one score significantly (noun-verb agreement) while the syntactic treatment reported significant changes in four sentence types. 3.3. Focus B: Cross-language transfer (CLT) of therapy outcomes This section includes studies that provided treatment in L2 or L1 and examined pre-post treatment measures in the untrained language (L1 and L2 respectively) for either receptive (questions 3&4; see Table 3) or expressive (questions 5&6; see Table 4) language skills. Table 3a shows that, of the five studies that examined CLT from L2 to L1, three showed significant improvement in receptive language skills (Faroqi & Chengappa 1996; Gil & Goral 2004; Khamis et al., 1996) while one study reported a post-treatment decline in performance (Miertsch et al., 2009). It is noteworthy that one of the studies with significant improvement involved chronic participants (Faroqi & Chengappa, 1996). Two studies, listed in Table 3b, reported significant CLT from L1 to L2 (Gil & Goral, 2004; Junque et al., 1989). The Junque et al., study (1989) is noteworthy because it examined 30 acutely aphasic bilingual participants and found significant improvement in single word identification. The magnitude of improvement in L2 was smaller than that observed in the treated L1. ---------------Insert Tables 3a and 3b about here--------------As for expressive language outcomes, there are data from 11 studies examining L2 to L1 and four studies reporting L1 to L2 (Table 4; Abutelabi et al., 2009; Edmonds & Kiran, 2006; Faroqi & Chengappa, 1996; Gil & Goral, 2004; Goral et al., 2009; Khamis et al., 1996; Marangolo et al., 2009; Meinzer et al., 2007; Penn & Beecham, 1992; and Ansaldo et al., 2009; Gil & Goral, 2004; Edmonds & Kiran, 2006; Junque et al., 1989 respectively). Five out of 11 studies found CLT to the untrained L1, of which three described acute participants and two described chronic participants (Faroqi & Chengappa 1996; Khamis et al., 1996; Marangolo et al., 2009; Miertsch et al., 2009). Four out of these five studies focused on word retrieval and one study focused on sentence production. All four studies (35 participants total; one chronic and 34 acute) that examined CLT to the untrained L2 after L1 treatment reported significant improvement (Ansaldo et al., 2009; Gil & Goral, 2009; Edmonds & Kiran, 2006; Junque et al., 1989). ---------------Insert Tables 4a and 4b about here--------------3.4. Participant and language variables Table 5 provides a detailed description of the 45 bilingual participants included across studies (35 male, 10 female; age range = 21 to 80 years). Eighty-four percent of the participants (38/45) exhibited stroke-induced aphasia with the remaining 16% presenting with other neurologic impairments (6 hematomas, 1 traumatic brain injury). Aphasia type was characterized as fluent (67%; 30/45), non-fluent (31%; 14/45) or mixed (2%; 1/45). Of those that reported severity, three exhibited severe aphasia, one moderate to severe aphasia, 19 moderate and 15 mild or mild to moderate aphasia. Time post onset ranged from 2 weeks to 8 years; however most participants (89%; 40/45) demonstrated acute aphasia. Comparing L2 and CLT expressive and receptive outcomes (Tables 2-4) with these participant variables revealed no systematic relationship of aphasia type, severity or time post onset. Data sets from both acute and chronic participants reported positive treatment outcomes in several aspects, likewise there were instances where both acute and chronic participants failed to show improvements. ---------------Insert Table 5 about here--------------Table 6 lists the language typologies and language histories of the participants in each study. Language typologies were determined using the World atlas of language structures (Haspelmath, Dryer, Gil, & Comrie, 2008). There were five studies in which L1 and L2 belonged to the same language family (Abutelabi et al., 2009; Faroqi & Chengappa, 1996; Junque et al., 1989; Khamis et al., 1996; Miertsch et al., 2009). Of these studies, CLT was reported in two out of three for receptive language (Faroqi & Chengappa, 1996; Junque et al., 1989) and in four out of five for expressive language (Faroqi & Chengappa, 1996; Junque et al., 1989; Khamis et al., 1996; Miertsch et al., 2009). Nine participants were late bilinguals (age of L2 acquisition ≥ 5 years) and the remaining 36 were early bilinguals (acquisition ≤ 5 years). Age of acquisition did not appear to impact L2 outcomes or CLT. Proficiency of language use was reported in the majority of studies. Eight-four percent (38/45) of participants reported equal proficiency in L1 and L2. ---------------Insert Table 6 about here--------------- 4. Discussion The purpose of this evidence-based review was to examine language outcomes of unilingual SLP treatments for bilingual individuals with aphasia. Eight clinical questions were established a priori to tease apart expressive and receptive outcomes in the trained language and CLT. Further analysis of patient-related, aphasia-related and linguistic aspects was conducted to determine what variables, if any, impact treatment outcomes. The results of the systematic search of literature from 1980 onwards yielded 14 exploratory studies from 13 citations pertaining to one or more of the clinical questions; the majority of which provided data pertaining to unilingual treatment in L2 on expressive language outcomes (N = 12). There was also a preponderance of studies of acutely aphasic patients. No studies were found allowing us to determine the impact of service provider (i.e. language broker) on treatment outcomes. Although the limited number and methodological quality of the included studies warrants caution when interpreting the results, this review revealed the following trends: therapy provided in L2 yields positive receptive and expressive outcomes even in chronic bilingual aphasia, CLT does occur in over half the participants, and, age of acquisition and language typology have little differential effect on outcomes. This EBSR also illuminated that several variables are confounded, and consequently need further research in order to clearly delineate their effects on bilingual aphasia outcomes. The findings of this review and recommendations for future research are discussed in the following paragraphs. 4.1. Clinical Implications SLPs face the challenge of making treatment decisions that will facilitate and optimize recovery of both languages of a bilingual aphasic individual. A recent survey of SLPs who worked with adults in the United States revealed that a majority felt that their academic and clinical training left them inadequately prepared for assessment and treatment of bilingual aphasic clients (Centeno, 2009). Further, SLPs expressed dissatisfaction with the amount of information available to guide treatment decisions. Given that a majority of the world (and a rapidly growing US demographic) is bilingual, Centeno’s (2009) survey presents the rather disturbing possibility that a significant proportion of the world’s aphasic clients’ communicative needs may be compromised due to a limited knowledge base. Hence, it is imperative to propose clinical recommendations based on this EBSR, bearing in mind the earlier cautionary note that in most cases, the evidence was only modest. The first clinically relevant finding of this EBSR is that all studies investigating the direct impact of unilingual treatment in L2 showed improvement across receptive and expressive language modalities. Given that the efficacy of aphasia treatment has been well-established (Robey, 1998; Wisenburn & Mahoney, 2009), this finding with bilingual aphasia is not surprising. It is recommended that when an SLP is faced with decisions about choice of language for treatment, the L2 can be actively considered as a viable option. Of course, factors such as client preferences and language of the environment should be considered. This EBSR failed to reveal any consistent effect of L2 acquisition age or proficiency level as is evident from the participant variables in Tables 5 and 6 (but see Edmonds & Kiran, 2006). Hence, until further data emerge suggesting otherwise, SLPs may consider L2 therapy for early and late bilinguals with moderate to high L2 proficiency. It should be stressed that these recommendations are not to be interpreted as L1 treatment is undesirable, rather, that L2 treatment does not seem to have any negative impact on outcomes (see also Kohnert, 2009 p.184 for a similar suggestion). The benefits of cross linguistic transfer are less clear from this EBSR. Some unilingual therapy studies found no generalization to the untrained language (Meinzer et al., 2007) while others reported generalization (Edmonds & Kiran, 2006; Gil & Goral, 2004; Miertsch et al., 2008). The recent review of 12 studies of bilingual aphasia treatment by Kohnert (2009) also found mixed CLT effects. These mixed CLT findings may be due to a plethora of factors such as differences between treatment approaches, focus on different language domains such as lexical retrieval or syntax, structural differences between languages, and patient-related variables such as pre- morbid language proficiency, type of aphasia, relative severity of impairment in each language, and extent/size of the neurological lesion. Interestingly, studies addressing receptive language appeared to show more positive cross linguistic effects. The implications for clinical decision making purposes then are that the current state of evidence does not provide any basis for SLPs to predict if CLT will occur after unilingual treatment. Some authors have suggested that the chances of CLT can potentially be increased by inclusion of activities such as translation between L1 and L2, or stimuli with structural overlap between L1-L2, and help from family members (Ansaldo et al., 2008; Kohnert, 2004; 2009). However, the studies included in the present EBSR are inadequate in validating these suggestions and further research is warranted. 4.2. Future research needs This EBSR revealed the rather appalling dearth of methodologically rigorous bilingual aphasia treatment studies. It also identified multiple questions for future research although an exhaustive list is beyond the scope of this paper. Foremost, at least to our knowledge, there is virtually no research on the effectiveness of language brokers. Hence two clinical questions could not be addressed in this EBSR. Given that language brokers are frequently utilized in treatment of bilingual aphasic clients (Centeno, 2009), research on the extent of and factors influencing outcomes with language brokers is crucial. The second major problem in the existing corpus of studies is the over representation of studies with acutely aphasic patients that failed to account for spontaneous neurological recovery. Future research will need to better delineate spontaneous versus treatment-induced neural plasticity. Treatment-specific factors such as effects of modality of treatment (expression, comprehension, reading), language domain (lexical, morphosyntactic etc.), cross-linguistic similarity on CLT, and dose-response (frequency/intensity) characteristics need to be examined. Other long recognized areas of research are the influence of L2 acquisition age, pre-morbid proficiency, and daily usage patterns. 4.3. Methodological considerations Bilingual aphasia treatment studies at minimum, differ in languages used, types of treatments used, types of outcome measures used, treatment schedules (intensity-frequency), and types of participants (acute versus chronic aphasia; early versus late bilinguals; lesion location and size). It is therefore difficult to delineate the impact of these variables on bilingual treatment outcomes and to compare effects across studies. A further hurdle in this EBSR was not necessarily the limited number of studies, but the limited methodological detail provided in some of the studies. Several studies failed to mention crucial aspects such as the presence of pathological code-switching, type of activities included in treatment, or the language of the home/community. With a few exceptions, the overall methodological rigor of bilingual treatment studies reviewed in this EBSR was below that of monolingual treatment studies of aphasia (e.g., compared to Cherney, Patterson, Raymer, Frymark, & Schooling, T., 2008). In light of these issues, we conclude this EBSR by presenting a checklist for authors who plan to publish bilingual aphasia treatment studies. This checklist is intended to recommend information that should be included in order to make each bilingual treatment study transparent, replicable, and interpretable. As the number of bilingual aphasic clients and hence the number of bilingual treatment studies proliferates, this recommendation is crucial for clear scientific communication that will aid future systematic reviews. 1. Research Questions. Akin to treatment studies of monolingual aphasic participants, a clear articulation of the rationale for the treatment chosen, the experimental questions, and the apriori hypotheses is warranted. This includes, but is not limited to, expectations of within and across language outcomes in various domains such as word retrieval and sentence formulation. 2. Experimental Design. The study design should be compatible with the experimental question, with adequate consideration of non-treatment variables that may confound treatment outcomes. These include, but are not limited to, neurological factors such as spontaneous recovery in the early months and psychosocial factors such as the nature of language stimulation outside the treatment setting and motivation to improve in a specific language. Internal and external validity needs to be demonstrated (Thompson & Kearns, 1991). Given the importance of massed practice (that is, high intensity of treatment) on aphasia therapy outcomes, it is imperative that authors justify their choice of treatment dosage (Cherney et al., 2008; Kleim & Jones, 2008). 3. Participants. Participant(s)’ neurological, cognitive, linguistic, and demographic profiles need to be explicit enough to enable comparison across studies. Details of pre-morbid and post-morbid language background and language use history can be provided either with a custom-made questionnaire or by using published questionnaires (Marian et al., 2007; Munoz, Marquart, & Copeland, 1999; Paradis, 1989). Patterns of cognitive and linguistic deficits, including relative severity differences across languages and domain general therapy predictors (attention and (working) memory span), should be described. It has been pointed out that aphasic symptoms could manifest differently across languages because of typological differences. Studies examining CLT should specifically include data on translation abilities, pathological language mixing, and the presence of bilingual stimulation in the home/community environment. Description of the brain lesion should be in sufficient detail to enable future examination of cross-study associations with cognitive-linguistic deficit patterns, recovery patterns, and recovery predictors. This may be achieved by a detailed narrative of radiological findings and/or inclusion of structural magnetic resonance images (sMRI)4. If, as suggested by Ansaldo et al. (2008), lesion location needs to be considered while choosing appropriate therapy for the bilingual client (p. 554), lesion information is vital for future therapy decisions. 4. Treatment protocol and outcome measures. The treatment protocol should be in adequate detail to be replicable, with specific information about the language of treatment, feedback, choice of stimuli (cognate, phonological or syntactic overlap between languages), treatment intensity-frequency, and the person administering the treatment (monolingual, bilingual). It is highly recommended that outcome measures include published tests to enable comparison across studies, as well as (custom-made) tests that will demonstrate the direct effects of the treatment on trained and untrained domains. One needs to be cognizant of the fact that researchers should report all measures, irrespective of whether these change significantly after treatment (e.g., Abutelabi et al., 2009). 5. Interpretation. The results should be interpreted cautiously and within the context of potential sources of bias, whether they arise from the study design, measurement imprecisions, participant inconsistencies, or statistical analyses. Whenever possible, authors should discuss the relative influence of bilingual variables (age of acquisition, 4 We thank an anonymous reviewer for highlighting the importance of including sMRI information. level of proficiency, acceptability of code-mixing among unimpaired bilinguals in the community), linguistic variables (especially similarities between languages), aphasiological variables (including severity, deficit pattern, pathological code-mixing, and cognitive aspects), neurological variables (lesion site-extent including subcortical lesions and white matter involvement, multiple strokes), and sociocultural aspects on treatment outcomes. 5. Conclusions In conclusion, although modest evidence exists for positive treatment outcomes in L2 and for CLT in bilingual aphasia, the results of this EBSR should be considered preliminary. Until further data on bilingual treatment emerge, treatment decisions can be made based on the findings of this EBSR complemented with clinical expertise, client preferences, and consideration of sociocultural variables. It is evident from limited number of published bilingual aphasia treatment studies that this research enterprise is still in its infancy and in need of considerable systematic research. Investigations of bilingual aphasia treatment outcomes are necessary not only for its obvious clinical importance, but also for the insights gleaned about language representation and breakdown in the bilingual brain. Lesion-deficit correlations hold the potential for refining neuroanatomical models of bilingual language representation. Associations between language recovery and lesion characteristics (as well as cognitive linguistic deficits) will contribute to our understanding of rehabilitation-dependent neural plasticity. Appendix A.1 List of Electronic Databases Searched AgeLine The Aphasiology Archive CINAHL Cochrane Library ComDisDome Communications & Mass Media Complete CSA Linguistics Language Behaviour Abstracts CSA Neurosciences Abstracts CSA Social Services Abstracts Education Research Complete ERIC Evidence-Based Medicine Guidelines GoogleScholar Health Source: Nursing HighWire Press Latin America and Caribbean Health Sciences Literature NHS Evidence: Health Information Resources PsycBITE PsycINFO Psychology and Behavioral Sciences Collection PubMed REHABDATA ResearchGATE Science Citation Index ScienceDirect Social Science Citation Index speechBITE SUMSearch TRIP Database A.2 Additional Searches The reference lists of all relevant articles identified were scanned for other possible studies. All ASHA journals were searched through the HighWire Press website. Hand-search of International Journal of Bilingualism and the Journal of Multilingual Communication Disorders. 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Analytical and Technical Directions in Applied Aphasia Analysis: The Midas Touch. In Clinical Aphasiology Conference: Clinical Aphasiology Conference (1989: 19th : Lake Tahoe, NV : June 1989) /: Pro-Ed(1991), pages 41-54. Watamori, T., & Sasanuma, R. (1978). The recovery processes of two English-Japanese bilingual aphasics. Brain and Language, 6, 127-140. Wisenburn, B., & Mahoney, K. (2009). A meta-analysis of word-finding treatments for aphasia. Aphasiology, 23, 1338-1352. * Studies that were included in the EBSR Figure Caption: Figure 1. Flowchart outlining the study identification process Table 1. Studies Included in EBSR by Questions Addressed, Quality Markers, and Number of Participants. Quality indicators Citation Abutalebi, et al., 2009 Ansaldo et al., 2009 Question(s) N Study Design Protocol Description 1,2,3,5 1 Case study + Convenience - - - Case study + Convenience - + - 6 1 Sampling Treatment Fidelity Significance Precision Edmonds & Kiran, 2006 2,5,6 3 Single subject + Convenience + + + Faroqi & Chengappa, 1996 1-3,5 1 Case study + Convenience - - - Gil & Goral, 2004 1-6 1 Case study + Convenience - - - Goral, et al., 2009 2,5 1 Case study + Convenience - - +/- Junqué et al., 1989 4,6 30 Case series + Convenience - + - Khamis et al.,1996 1,2,3,5 1 Case study + Convenience - - - 2 2 Single subject + Convenience + + - Maragnolo et al., 2009 (Study 1) 2,5 1 Case study + Convenience - + - Maragnolo et al., 2009 (Study 2) 2,5 1 Case study + Convenience - + + Meinzer et al., 2007 2,5 1 Case study + Convenience - - - Miertsch et al., 2009 1,2,3,5 1 Case study - Convenience - + + 2,5 1 Case study + Convenience - - - Laganaro et al., 2003 Penn & Beecham, 1992 Note. + = Present; - = Absent Table 2a. Outcomes of SLP Treatment in L2 on Receptive Language in L2 (Question 1) Citation Abutalebi et al., 2009 Intervention Word-phonology during Phase 1 Word- phonology + Wordsemantics during Phase 2 Faroqi & Chengappa 1996 Gil & Goral 2004 Khamis et al., 1996 Miertsch et al., 20091 Sentence-syntax Auditory-comprehension Treatment Schedule Phase 1: 60 minute sessions 7 times weekly 6 weeks Phase 2: 60 minute sessions 4 times weekly 16 weeks 60 minute sessions 32 sessions Outcome(s) Measured BAT Pointing Commands Verbal-auditory discrimination Syntactic comprehension Lexical decision Listening comprehension Grammatical judgment: Active sentences Passive sentences Object-clefts Wh-questions Relative clauses Comprehension: Active sentences Passive sentences Object-clefts Wh-questions Relative clauses Pre Post 9/10 5/10 9/18 76/86 24/30 4/5 10/10 10/10 18/18* 85/86* 30/30* 5/5 64% 60% 60% 72% 52% 100%* 100%* 98%* 100%* 96%* 75% 46% 40% 62% 66% 100%* 98%* 88%* 100%* 90%* 45 minute sessions 5 times weekly 4 weeks ILAT Answering questions Picture identification Commands NR 42% 40% NR 87%* 55%* Multiple: auditorycomprehension + word + discourse strategies 8 weeks ILAT Picture identification Commands 92% 99% 100%* 100% Word-semantic 45 minute sessions 2 hours daily 22 sessions 50% 100%* BAT Syntactic comprehension of reversible noun phrases Note. AAT=Aechan Aphasia Test; BAT=Bilingual Aphasia Test; ILAT= Israeli Lowenstein Aphasia Test; *McNemar’s change test, p<0.05 1 This study examined the effect of L3 treatment on L1, L2, and L3 Table 2b. Outcomes of SLP Treatment in L2 on Expressive Language in L2 (Question 2) Citation Abutalebi et al., 2009 Intervention Treatment Schedule Word-phonology during Phase 1 Phase 1: 60 minute sessions 7 times weekly 6 weeks Phase 2: 60 minute sessions 4 times weekly 16 weeks BAT Synonyms Antonyms Word repetition Sentence repetition Series Naming Semantic opposites SNB 2 hour sessions 2 times weekly BNT BAT Naming Word Repetition Semantic Categories Semantic opposites Synonyms Antonyms I Antonyms II 60 minute sessions 32 sessions Word- phonology + Wordsemantics during Phase 2 Edmonds & Kiran, 2006 Participant 2 Faroqi & Chengappa, 1996 Gil & Goral, 2004 Goral et al., 2009 Word-semantic Sentence-syntax Auditory-comprehension Sentence-syntax Outcome(s) Measured Pre Post 8/20 8/10 30/30 7/7 0/3 8/20 8/10 57/144 19/20* 10/10 30/30 7/7 3/3 19/20* 10/10 122/144* 2/60 35/60* 0% 77% 60% 0% 20% 20% 20% 43%* 73% 80%* 70%* 100%* 60%* 40%* Wh-marker production Who What When Where 40% 30% 40% 20% 100%* 100%* 100%* 100%* 45 minute sessions 5 times weekly 1 month ILAT Spontaneous speech Elicited speech Repetition Naming 30% 0% 51% 8% 30% 0% 69%* 20%* 60 minute sessions Treatment A 3 times weekly 9 sessions Khamis et al., 1996 Lagarno et al., 2003 Multiple: auditorycomprehension + word + discourse strategies 8 weeks Word-phonology Daily sessions 2 week intervals 6 weeks 60 minute sessions 5 times weekly 6 months Noun-verb agr Person-gender agr Tense consistency Preposition use Syllables per minute Treatment B Noun-verb agreement Person-gender agreement Tense consistency Preposition use Syllables per minute ILAT Spontaneous speech Automatic speech Naming 57% 91% 85% 90% 38 73%* 100% 88% 93% 46 72% 95% 83% 72% 39 67% 93% 80% 85%* 40 80% 100% 50% 90%* 100% 70%* 4% 14% 20% 40%* 24%* 45%* 53% 73% 62% 70%* 83%* 66%* AAT Repetition Naming 46% 60% 52%* 69%* Naming Participant 7 Computerized treatment items Behavioral treatment items Control items Participant 10 Computerized treatment items Behavioral treatment items Control items Marangolo et al., 2009 (Study 1) Word-phonology Marangolo et al., 2009 (Study 2) Word-phonology 2 hour sessions 5 times weekly 2 weeks Naming 35/48 47/48* Meinzer et al., 2007 Word-semantic 3 hours daily 10 consecutive days Naming 38/80 54/80* Miertsch et al., 20091 Word-semantic 45 minute sessions 2 hours daily BAT Repetition of sentences 1/8 7/8* 22 sessions Penn & Beecham 1992 Sentence-syntax 9 sessions 14 weeks Words per minute 130 115 Compensatory strategy use: + Circumlocution + Fluency place holder + Turn taking + Prenominalization + Note. AAT=Aechan Aphasia Test; BAT=Bilingual Aphasia Test; BNT=Boston Naming Test; ILAT= Israeli Lowenstein Aphasia Test; SNB=Snodgrass Naming Battery; * McNemar’s change test, p<0.05; + = Present; - = Absent 1 This study examined the effect of L3 treatment on L1, L2, and L3. Table 3a. CLT of SLP Treatment in L2 on Receptive Language in L1 (Question 3) Citation Abutalebi et al., 2009 Intervention Word-phonology during Phase 1 Word- phonology + Wordsemantics during Phase 2 Faroqi & Chengappa, 1996 Gil & Goral, 2004 Khamis et al., 1996 Miertsch et al., 20091 Sentence-syntax Auditory-comprehension Treatment Schedule Phase 1: 60 minute sessions 7 times weekly 6 weeks Phase 2: 60 minute sessions 4 times weekly 16 weeks 60 minute sessions 32 sessions Outcome(s) Measured BAT Pointing Commands Verbal-auditory discrimination Syntactic comprehension Lexical decision Listening comprehension Grammatical judgment: Active sentences Passive sentences Object-clefts Wh-questions Relative clauses Comprehension: Active sentences Passive sentences Object-clefts Wh-questions Relative clauses Pre Post 9/10 10/10 8/18 85/86 28/30 5/5 8/10 10/10 10/18 80/86 26/30 5/5 70% 54% 46% 64% 46% 100%* 100%* 92%* 92%* 84%* 56% 54% 44% 65% 54% 92%* 90%* 82%* 88%* 78%* 45 minute sessions 5 times weekly 4 weeks ILAT Answering questions Picture Identification Commands Reading 20% 44% 48% 13% 53%* 98%* 65%* 48%* Multiple: auditorycomprehension + word + discourse strategies 8 weeks ILAT Picture identification Commands 93% 92% 100%* 100%* Word–semantic 45 minute sessions 2 hours daily 22 sessions 80% 60%* Note. BAT=Bilingual Aphasia Test; ILAT=Israeli Lowenstein Aphasia Test *McNemar’s change test, p<0.05 1 This study examined the effect of L3 treatment on L1, L2, and L3. BAT Syntactic comprehension of reversible noun phrases Table 3b. CLT of SLP Treatment in L1 on Receptive Language in L2 (Question 4) Citation Gil & Goral, 2004 Junque et al., 1989 Intervention Auditory–comprehension Unspecified Note. ILAT=Israeli Lowenstein Aphasia Test; NR = not reported * McNemar’s change test, p<0.05 a t-test as reported by study authors (N=30) Treatment Schedule 45 minutes 5 times weekly 6 weeks NR Outcome(s) Measured ILAT: Commands Picture identification Reading Object identification Pre Post 95% 80% 60% 92% 95%* 75%* 19.97% 22.47%a Table 4a. CLT of SLP Treatment in L2 on Expressive Language in L1 (Question 5) Citation Abutalebi et al., 2009 Intervention Treatment Schedule Word-phonology during Phase 1 Phase 1: 60 minute sessions 7 times weekly 6 weeks Phase 2: 60 minute sessions 4 times weekly 16 weeks BAT Synonyms Antonyms Word repetition Sentence repetition Series Naming Semantic opposites SNB 2 hour sessions 2 times weekly BNT BAT Naming Word repetition Semantic categories Semantic opposites Synonyms Antonyms I Antonyms II 60 minute sessions 32 sessions Auditorycomprehension Sentence-syntax Word- phonology + Word-semantics during Phase 2 Edmonds & Kiran, 2006 Participant 2 Faroqi & Chengappa, 1996 Gil & Goral, 2004 Goral et al., 2009 Word-semantic Sentence- syntax Outcome(s) Measured Pre Post 4/5 4/5 30/30 7/7 3/3 9/20 7/10 35/90 4/5 3/5 30/30 7/7 3/3 8/20 7/10 34/90 0/60 0/60 0% 67% 100% 10% 20% 0% 60% 0% 67% 100% 10% 20% 0% 60% Wh-marker production who what when where 30% 30% 40% 20% 100%* 100%* 100%* 90%* 45 minute sessions 5 times weekly 1 month ILAT Spontaneous speech Elicited speech Repetition Naming 0% 51% 4% 19% 0% 51% 4% 19% 60 minute sessions 3 times weekly 9 sessions Treatment A Noun-verb agr Person-gender agr 97% 100% 97% 100% Khamis et al., 1996 Tense consistency Preposition use Syllables per minute Treatment B Noun-verb agr Person-gender agr Tense consistency Preposition use Syllables per minute 99% 93% 74 99% 96% 76 97% 100% 97% 97% 78 98% 100% 98% 96% 73 ILAT Spontaneous speech Automatic speech Naming 80% 100% 53% 90%* 100% 79%* 44 61 49 67* Multiple: auditorycomprehension + word + discourse strategies 8 weeks Marangolo et al., 2009 (Study 1) Word-phonology 60 minute sessions 5 times weekly 6 months Marangolo et al., 2009 (Study 2) Word-phonology 2 hour sessions 5 times weekly 2 weeks Naming 27/48 46/48* Meinzer et al., 2007 Word-semantic 3 hours daily 10 consecutive days Naming 4/80 4/80 Miertsch et al., 20091 Word-semantics 45 minute sessions 2 hours daily 22 sessions BAT Repetition of sentences 50% 81%* Penn & Beecham, 1992 Sentence-syntax 9 sessions 14 weeks AAT Repetition Naming Compensatory strategy use: Circumlocution Fluency place holder Turn taking Prenominalization Note. AAT=Aechan Aphasia Test; BAT=Bilingual Aphasia Test; BNT=Boston Naming Test; ILAT= Israeli Lowenstein Aphasia Test; SNB=Snodgrass Naming Battery; + = Present; - = Absent *McNemar’s change test, p<0.05; 1 This study examined the effect of L3 treatment on L1, L2, and L3. Table 4b. CLT of SLP Treatment in L1 on Expressive Language in L2 (Question 6) Citation Ansaldo et al., 2009 Gil & Goral, 2004 Edmonds & Kiran, 2006 Participant 1 Participant 2 Participant 3 Intervention Treatment Schedule Discourse strategies + Word–semantic 1 hour sessions 2 times weekly 3 months Auditorycomprehension 45 minute sessions 5 times weekly 1 month Word–semantic 2 hour sessions 2 times weekly Word–semantic Word-semantic 2 hour sessions 2 times weekly 2 hour sessions 2 times weekly Outcome(s) Measured WAB Repetition Object naming Sentence completion Responsive speech ILAT Repetition Elicited speech Naming Writing BNT BAT Naming Word repetition Semantic categories Semantic opposites Synonyms Antonyms I Antonyms II BNT BAT Naming Word repetition Semantic categories Semantic opposites Synonyms Antonyms I Antonyms II BNT BAT Naming Pre Post 84/100 30/60 6/10 4/10 90/100* 47/60* 7/10 7/10 74% 5% 31% 20% 96%* 40%* 51%* 50%* 41/60 48/60* 60% 93% 100% 20% 80% 80% 60% 62% 97% 80%* 40%* 80% 80% 100%* 2/60 35/60* 0% 77% 60% 0% 20% 20% 20% 41%* 73% 80%* 70%* 100%* 60%* 40%* 23/60 33/60* 88% 95%* Word repetition Semantic categories Semantic opposites Synonyms Antonyms I Antonyms II Junque et al., 1989 Unspecified NR Naming 57% 100% 30% 20% 40% 40% 77%* 60%* 20%* 0%* 60%* 0%* 9.70 13.67a Note. AAT=Aechan Aphasia Test; BAT=Bilingual Aphasia Test; BNT=Boston Naming Test; ILAT=Israeli Lowenstein Aphasia Test ; NR = not reported; WAB=Western Aphasia Battery *McNemar’s change test, p<0.05 a t-test as reported by study authors (N=30) Table 5. Participant variables Aphasia Type Fluent Aphasia Severity NR TPO Chronic (2y) L CVA Subcortical, including basal ganglia and internal capsule Fluent Severe Acute (2m) 10-12y L CVA NR Non fluent 2 Moderate 1 Severe Acute (8-9m) F 17y L CVA Inferior frontal and basal ganglia Non fluent NR Chronic (15m) 57y M >18y L CVA Fronto-parietal lobes Mixed Severe Acute (2w4m) 1 49y M >18y L CVA Fronto-temporo-parietal lobes Non fluent NR Chronic (7y) Junque et al., 1989 30 M=60y SD=9.9y R=33-79y 23M 7F NR 24 CVA; 6 H NR 22 Fluent 8 Non fluent 17 Moderate 13 Mild Acute (1-6m) Khamis et al., 1993 1 21y M NR TBI Frontal lobe Fluent NR Acute (2m) Laganaro et al., 2003 2 P7=80y P10=69y 2M NR L CVA NR Fluent Moderate to severe Acute (NR) Maragnolo et al., 2009 1 60y F NR L CVA Fluent NR Acute (2m) Citation Ansaldo et al., 2009 N 1 Age 56y Gender M Education >18y Etiology/Site of lesion L CVA Subcortical including basal ganglia Abutalebi et al., 2009 1 56y M 12y Edmonds & Kiran, 2006 3 P1=53y P2=53y P3=56y 2M 1F Faroqi & Chengapppa, 1996 1 31y Gil & Goral, 2004 1 Goral et al., 2009 Temporo-parietal, incl., STG, MTG, SMG Meinzer et al., 2007 1 35y M >18y L CVA Fronto-temporal-parietal lobes Non fluent Severe Chronic (32m) Miersch et al., 2009 1 48y M >18y L CVA Temporal lobe Fluent Mild to moderate Chronic (8y) Penn & Beecham, 1992 1 38y M >18y L CVA Parietal lobe Fluent Mild Acute (9m) Note. F=Female; H=Hematoma; L CVA=Left cerebrovascular accident; M=Male; M=Mean’ m=Months; MTG= Middle Temporal Gyrus; N= participant Number; NR=Not reported; P=Participant; R=Range; SD=Standard deviation; SMG= SupraMarginal Gyrus; STG=Superior Temporal Gyrus; TPO =Time post onset; w=Weeks; y=Years Table 6. Language variables Citation L1 L2 L2 Acquisition L1 Proficiency Level L2 Proficiency Level Ansaldo et al., 2009 Spanish IE>Rom English IE>Ger Late High High Abutalebi et al., 2009 Spanish IE>Rom Italian IE>Ro Late NR High Edmonds & Kiran, 2006 Spanish IE>Rom English IE>Ger P1 Late P2 Early P3 Early P1 High P2 Low P3 Low P1 High P2 High P3 High Telugu Dravidian>SouthCen Kannada Dravidian>South Early High High Gil & Goral, 2004 Russian IE>Sla Hebrew AA>Sem Late High High Goral et al., 2009 Hebrew AA>Sem English IE>Ger Early High High Junque et al., 1989 Catalan IE>Rom Spanish IE>Rom Early High High Khamis et al. 1993 Arabic AA>Sem Hebrew AA>Sem Late High High Laganaro et al., 2003 NR French IE>Rom Late NR High Marangolo et al., 2009 Flemish IE>Ger Italian IE>Rom Late High High Meinzer et al., 2007 French IE>Rom German IE>Ger Early High High Meirtch et al., 2009 German IE>Ger English (L2) IE>Ger French (L3) IE>Rom Late NR High Penn & Beecham, 1992 Bantu NC>Ban English IE>Ger Late High NR Faroqi & Chengapppa, 1996 Note. AA=AfroAsiatic; Ban=Bantoid; Early= < age 5; Ger=Germanic; IE=IndoEuroprean; Late= > age 5; NC=NigerCongo; NR=Note reported; Rom=Romance; Sem=Semitic; Sla=Slavic; South=Southern; SouthCen=SouthCentral Supplementary Materials S1. Expanded Search Term ("Multilingualism"[Mesh] OR "Emigrants and Immigrants"[Mesh]) AND "Aphasia"[Mesh] ("Multilingualism"[Mesh] OR "Emigrants and Immigrants"[Mesh]) AND ("Language Disorders/rehabilitation"[Mesh] OR "Speech Disorders/rehabilitation"[Mesh] OR "Rehabilitation of Speech and Language Disorders"[Mesh]) AND ("Brain Injuries"[Mesh] OR ―Cerebrovascular Disorders‖[Mesh] OR ―Dementia‖[Mesh]) ("Multilingualism"[Mesh] OR "Emigrants and Immigrants"[Mesh]) AND ("Language Disorders/rehabilitation"[Mesh] OR "Speech Disorders/rehabilitation"[Mesh] OR "Rehabilitation of Speech and Language Disorders"[Mesh]) ("Multilingualism"[Mesh] OR "Emigrants and Immigrants"[Mesh]) AND ("Brain Injuries"[Mesh] OR ―Cerebrovascular Disorders‖[Mesh] OR ―Dementia‖[Mesh]) "Aphasia/rehabilitation"[Mesh] AND (multilingual* OR bilingual* OR trilingual* OR (english language) OR l1 OR l2 OR second* OR (cross linguistic) OR (cross language)) "Aphasia/rehabilitation"[Mesh] AND (multilingual* OR bilingual* OR trilingual* OR l1 OR l2 OR ("cross linguistic") OR ("cross language") OR ("limited proficiency") OR second*) ("Aphasia"[Mesh] OR "Anomia"[Mesh]) AND (multilingual* OR bilingual* OR trilingual* OR l1 OR l2 OR ("cross linguistic") OR ("cross language") OR ("limited proficiency") OR second* OR dual OR broker OR interpreter OR transfer) "Multilingualism"[MAJR] AND (interpreter OR broker) aphasia AND (*lingual* OR l1 OR l2 OR ell OR dual OR second* OR proficien*) AND (language AND (treatment OR therapy OR rehabilitation)) ((bilingual* OR multilingual* OR trilingual* OR polyglot OR (english language learner) OR (dual language) OR (l1) OR (l2)) AND aphasia) AND (treatment OR therapy OR rehabilitation OR intervention) "Aphasia"[Mesh] AND "Language"[Mesh] AND ("Reading"[Mesh] OR ―Speech‖[Mesh] OR ―Translating‖[Mesh] OR ―Writing‖[Mesh] OR ―Phonetics‖[Mesh] OR ―Semantics‖[Mesh] OR ―Vocabulary‖[Mesh]) AND (treatment OR therapy OR rehabilitation) ("Rehabilitation of Speech and Language Disorders"[Mesh] OR "Language Disorders/rehabilitation"[Mesh] OR "Speech Disorders/rehabilitation"[Mesh]) AND ("Aphasia"[Mesh] OR "Anomia"[Mesh]) (MM "Aphasia+") AND (MH "Multilingualism") Aphasi* AND (interpreter OR broker) "multilingual" ((MM "Multilingualism") OR (MH ―Multilingualism‖)) AND ((MM "Aphasia+") OR (MH ―Aphasia+‖)) ((XX "aphasia") OR (XX "aphasic")) AND (multilingual* OR bilingual* OR trilingual* OR polyglot OR (english language learner)) DE "BILINGUALISM" AND DE "APHASIA" (KW "multilingual/multicultural group" OR KW "multilingual" OR KW "bilingual") AND (KW "aphasia") ((ZW "bilingual") or (ZW "bilingual aphasia") or (ZW "bilingualism") or (ZW "bilinguals") or (ZW "multilingual") or (ZW "multilingual/multicultural group") or (ZW "multilingualism") or (ZW "english language learners")) and ((ZW "aphasia") or (ZW "aphasia treatment")) (DE "APHASIA" OR DE "AGRAMMATISM" OR DE "ANOMIA" OR DE "CONDUCTION aphasia" OR DE "JARGON aphasia" OR DE "WORD deafness") and (DE "MULTILINGUAL persons" OR DE "MULTILINGUALISM" OR DE "BILINGUALISM" OR DE "MULTICULTURALISM") ((ZU "aphasia") or (ZU "aphasia -- treatment") or (ZU "aphasic persons")) and ((ZU "multilingual persons") or (ZU "multilingualism") or (ZU "bilingualism")) (DE "Aphasia" OR DE "Acalculia" OR DE "Agnosia" OR DE "Agraphia" OR DE "Dysphasia") and (DE "Multilingualism" OR DE "Bilingualism") (DE "APHASIA") AND (DE "BILINGUALISM" OR DE "CODE switching (Linguistics)" OR DE "EDUCATION, Bilingual" OR DE "INTERFERENCE (Linguistics)" OR DE "LANGUAGE attrition" OR DE "MULTILINGUALISM") (DE "APHASIA") AND (DE "BILINGUALISM" OR DE "MULTILINGUALISM") DE=("agnosia" or "aphasia" or "traumatic brain injury tbi") and ("bilingualism") DE=("multilingualism" or "bilingualism" or "cultural background") and("speech therapy" or "language therapy") and ("agnosia" or "aphasia" or "traumatic brain injury tbi") DE=("aphasia") and ("bilingualism" or "code switching" or "cross cultural communication" or "diglossia" or "indigenous languages" or "language contact" or "language diversity" or "language proficiency" or "language use" or "languages" or "multilingualism" or "second language learning" or "second languages" or "social factors" or "sociolinguistics" or "sprachbund") DE=("aphasia") and ("bilingualism" or "code switching" or "cross cultural communication" or "diglossia" or "indigenous languages" or "language contact" or "language diversity" or "language proficiency" or "language use" or "languages" or "multilingualism" or "second language learning" or "second languages" or "social factors" or "sociolinguistics" or "sprachbund") and ("speech therapy" or "therapy" or ―language therapy‖) DE=aphasia and (bilingual* or multilingual* or polyglot or (english language learner) or (dual language) or (l1) or (l2) or (cross linguistic)) TS=(((bilingual* OR multilingual* OR trilingual* OR polyglot OR (english language learner) OR (dual language) OR (l1) OR (l2) OR (cross linguistic)) AND aphasia) AND (treatment OR therapy OR rehabilitation OR intervention)) AND Language=(English) DE=‖aphasia‖ DE=‖aphasia‖ AND (DE=‖bilingualism‖ or DE=‖multilingualism‖ or DE=‖English (Second Language)‖ or DE=‖Second Language Learning‖) ("BILINGUAL" or "BILINGUALISM" or "BILINGUALS" or "MULTILINGUAL" or "MULTILINGUALISM" or "POLYGLOT" or "CROSS-CULTURAL") and ("APHASIA" or "APHASIC" or "APHASICS" or "APHASIOLOGICAL" or "APHASIOLOGY") (aphasia OR aphasic) AND (bilingual OR multilingual OR trilingual OR multicultural) (aphasia OR aphasic) AND (polyglot OR (english language learner) OR (dual language) OR cross) Supplementary Materials S2. 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Blinding not stated The procedure used to ensure that the treatment protocol is delivered as intended. The likelihood that the study findings occurred by chance. Evidence of treatment fidelity. No evidence of treatment fidelity Precision The size or magnitude of any difference found between the treatment under investigation and the control condition. Effect size and confidence interval reported or calculable. Effect size or confidence interval reported or calculable. Neither effect size nor confidence interval reported or calculable. Intention to treat Participants in a randomized controlled trial are analyzed according to the group to which they were initially assigned, regardless of whether or not they dropped out, fully complied to the treatment or crossed over and received other treatment. Analyzed by intention to treat. Not analyzed by intention to treat. Italics indicates highest quality marker Sampling/allocation Treatment fidelity Significance S3.1 Quality indicators used to evaluate included studies Random sample adequately described. Random sample inadequately described. Convenience/hand-picked sample. Not stated. P values reported or calculable. P values not reported or calculable. S3.2 State of research Exploratory research: Treatment approaches are developed and assessed in the context of whether they show promise of being efficacious. Efficacy research: Treatment approaches are rigorously tested under ideal, highly controlled conditions to determine the outcomes that result. Effectiveness research: If an intervention demonstrates positive outcomes in the highly controlled setting of a clinical trial, then the controls are relaxed to test the intervention in a ―real-world‖ clinical setting. Cost-benefit and/or public policy: Once an intervention has been shown to be both efficacious and effective, research is conducted to study the political and economic environment in which the intervention is best delivered. S3.3 Supplementary material reference S3 ASHA Leader [homepage on the Internet]. Rockville (MD): American Speech-LanguageHearing Association; c1997-2009 [2007 Mar 12; cited 2009 August 31]. Mullen R. The state of the evidence: ASHA develops levels of evidence for communication sciences and disorders. Available from http://www.asha.org/Publications/leader/2007/070306/f070306b.htm.
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