Original Article Risk Factors, Symptoms, and Treatment of Ovarian Torsion in Children: The Twelve-Year Experience of One Center Ziv Tsafrir, MD*, Foad Azem, MD, Joseph Hasson, MD, Efrat Solomon, BSc, Benny Almog, MD, Hagith Nagar, MD, Joseph B. Lessing, MD, and Ishai Levin, MD From the Department of Gynecology (Drs. Tsafrir, Azem, Hasson, Lessing, Almog and Levin), Lis Maternity Hospital, and the Department of Pediatric Surgery (Dr. Nagar and Ms. Soloman), Dana Children’s Hospital, Tel-Aviv Sourasky Medical Center, Sackler Faculty of Medicine, Tel-Aviv University, Tel Aviv, Israel. ABSTRACT Objective: To assess risk factors, clinical findings and mode of diagnosis and treatment in premenarchal children with surgically verified ovarian torsion (OT). Study Design: A retrospective case review (Canadian Task Force Classification II-2). Setting: Teaching and research hospital, a tertiary center. Patients: Premenarchal children with surgically verified OT. Interventions: Patients underwent either laparoscopy or laparotomy. Results: Twenty-two cases of OT in 20 premenarchal girls (median age 12 years) were identified. Three cases involved recurrent torsion after detorsion without cystectomy. The main presenting symptoms were sudden pain and vomiting. Six patients underwent Doppler examinations, and all demonstrated an abnormal flow. Seventeen interventions were by laparoscopy. Conservative management, mainly detorsion with additional cyst drainage or cystectomy, was performed in 19 cases (86.4%). Oophoropexy was performed in 3 cases (13.6%). Pathologic examination demonstrated 5 simple cysts and 1 dermoid cyst. Conclusions: Ovarian torsion in premenarchal girls usually presents with intermittent abdominal pain and abdominal tenderness. Other signs and symptoms are nonspecific. When performed, Doppler imaging may assist in diagnosing ovarian torsion in children. Detorsion followed by cystectomy may prevent recurrence. Journal of Minimally Invasive Gynecology (2012) 19, 29–33 Ó 2012 AAGL. All rights reserved. Keywords: DISCUSS Doppler; Ovarian torsion; Premenarchal treatment You can discuss this article with its authors and with other AAGL members at http://www.AAGL.org/jmig-19-1-11-00285 Use your Smartphone to scan this QR code and connect to the discussion forum for this article now* * Download a free QR Code scanner by searching for ‘‘QR scanner’’ in your smartphone’s app store or app marketplace. Ovarian torsion (OT) rarely occurs in childhood. Its annual prevalence is estimated to be 4.9 per 100 000 females aged 1 to 20 years . Nevertheless, it should be considered in the differential diagnosis of every young girl who presents with acute abdominal or pelvic pain . The symptoms and signs of OT are nonspecific and can be related to other The authors have no commercial, proprietary, or financial interest in the products or companies described in this article. The essence of the project was presented at the 13th World Congress On Controversies In Obstetrics, Gynecology & Infertility, which was held in Berlin, Germany, November 4-7, 2010. Corresponding author: Ziv Tsafrir, MD, Lis Maternity Hospital, Tel Aviv Sourasky Medical Center, 6 Weizman Street, Tel Aviv 64239, Israel. E-mail: [email protected] Submitted June 15, 2011. Accepted for publication August 19, 2011. Available at www.sciencedirect.com and www.jmig.org 1553-4650/$ - see front matter Ó 2012 AAGL. All rights reserved. doi:10.1016/j.jmig.2011.08.722 clinical causes, for example, gynecologic, urologic, or gastrointestinal [3–5]. Abdominal ultrasonography (AUS) is the modality of choice in the evaluation of the young female pelvis, providing clear visualization of the pelvic organs with no exposure to radiation, in addition to being highly available . The demonstration of an ovarian mass or of ovarian enlargement by AUS in a child with abdominal pain must raise the suspicion of OT. The Doppler flow has also been recently studied for the purpose of assisting in the diagnosis of OT, but the results on its accuracy in detecting pathology have not been consistent . The preferred treatment in the event of a confirmed OT is still a matter of debate as well. Ovarian vitality in the setting of OT is dependent on the duration of time until the diagnosis is established, before necrosis occurs. A growing number of studies advocate conservative surgery (ie, mainly detorsion 30 with additional cyst drainage or cystectomy), but this approach mainly applies to the adult population [7–13]. Moreover, although laparoscopy followed by a detorsion procedure is the usual approach for treating these cases, the apprehension of overlooking malignancy continues to be a matter of concern [14,15]. The necessity of performing an oophoropexy is also a point of contention [16,17]. Several recent reports focused on OT in the pediatric population [5,6,15]; however, they did not stratify for premenarchal girls. The aim of this study was to delineate risk factors, symptoms, signs, and mode of treatment in premenarchal girls diagnosed as having OT. Materials and Methods We conducted a chart review of all cases of children with surgically verified OT who were treated in our institution From January 1997 through January 2008. The institutional ethics committee approved this retrospective study. Only premenarchal girls were selected for study. The medical records were evaluated for patients’ age, medical and gynecologic history, presenting symptoms and signs, physical examination findings, laboratory results and imaging studies. We calculated the duration of symptoms until admission to the emergency department (ED), the interval between ED admission to undergoing AUS, and the time period between ED admission and transport to the operating room. Data on the type of surgical approach used to visualize the ovary, surgical findings, surgical procedure for treatment of the OT, pathologic results, postoperative complications, and whether the index OT was a recurrence of a prior one were analyzed. Statistical Analysis The c2 test or the Fischer exact test was used to compare categorical variables as appropriate, and the t test was used to compare means of continuous variables. Distribution was reported as means and SDs for normally distributed data and as medians for nonparametric data. Statistical analysis was performed with SPSS statistical software (IBM Corp., Armonk, New York). Results A total of 22 cases of OT in 20 premenarchal patients were identified. Their median age was 12 years (range 3 months to 16 years). The index OT was a recurrence of a previous one in 3 cases. The presenting symptom of abdominal pain was of abrupt onset in 17 of 22 cases, but all patients complained of intermittent pain, which was localized mostly to the lower abdomen. Vomiting during the painful episode was present as well (Table 1). The median time interval from pain onset to ER admission was 15 hours. Physical examination revealed abdominal tenderness in all but 1 case. There were positive peritoneal signs in 6 cases. High fever (.38.0 C, orally) was measured in 2 cases, and leukocytes (.11 ! 103/mL) were elevated in Journal of Minimally Invasive Gynecology, Vol 19, No 1, January/February 2012 Table 1 Signs and symptoms of ovarian torsion (20 patients, 22 cases) Sign/symptom Abdominal pain Abrupt onset Gradual onset Location of abdominal pain RLQ LLQ LA Diffuse Epigastric Vomiting Abdominal tenderness Peritoneal signs Fever R38 C WBCC .11 000/mL No. (%) of cases 17 (77.3%) 5 (22.7%) 9 (40.9%) 3 (13.7%) 5 (22.7%) 4 (18.2%) 1 (4.5%) 13 (59%) 21 (95%) 6 (27.3%) 2 (9%) 8 (36%) LLQ 5 Left lower quadrant; LA 5 lower abdomen; RLQ 5 right lower quadrant; WBCC 5 white blood cell count. 8 cases (36%). AUS with a full bladder had been performed in 20 cases and demonstrated enlarged ovaries (5–10 cm) in 8 cases and normal-sized ovaries with or without signs of edema in 8 and 4 cases, respectively. There was only 1 case of free fluid in the pelvis and no cases of complex cysts or abnormal mass. Doppler flow studies were performed in 6 cases, and the absence of blood flow to the affected ovary was demonstrated in all of them. Computed tomography examination was performed in 1 patient; however, this did not add any diagnostic information. The median time from ED admission to AUS imaging was 3.5 hours. The surgical approach to the affected ovary was laparoscopy in 17 cases, laparotomy in 3, and laparoscopy that converted to laparotomy in 2. Torsion was more common on the right side (68.2% vs 31.8% on the left side). The median number of adnexal twisting was 2 (range 1–6). The ovary was not enlarged in 13 cases (59.1%), whereas it was enlarged with or without a cyst (5–10 cm) in 5 cases (22.7%). The other 4 cases involved a paraovarian cyst. The median time interval from the ED to the operating room was 5.75 hours. The modes of therapeutic management are summarized in Table 2. Conservative management, mainly detorsion with additional cyst drainage or cystectomy, was the standard of care in most cases (19/22; 86.4%). Oophorectomy was performed because of 1 case each of excessive bleeding, a necrotic ovary, and a large dermoid cyst. Oophoropexy by plication of the ovarian ligament was performed in 3 cases, 2 of which were recurrences and 1 that was a prophylactic oophoropexy after contralateral oophorectomy. We had 3 cases of recurrent OT, 2 of which occurred after detorsion, and 1 after detorsion and cyst drainage. After surgery, 1 patient had bacteremia that was successfully treated with antibiotics. Tsafrir et al. Risk Factors, Symptoms, and Treatment of Ovarian Torsion in Children Table 2 Modes of treatment (20 patients, 22 cases) Treatment No. (%) of cases Detorsion Detorsion and drainage/fenestration Detorsion and cystectomy Detorsion and fixation* Ovarian resection Partial Complete 8 (36.4%) 6 (27.3%) 3 (13.6%) 3 (13.6%) 2 (9.1%) 1 (4.5%) * One case involved unilateral adnexectomy followed by fixation of the contralateral ovary. The pathologic study reports included simple cysts in 5 cases and 1 mature cystic teratoma (dermoid cyst). No other obvious pathologic findings were documented. The median hospital stay was 3 days. Discussion This retrospective chart study was conducted on a homogeneous group of premenarchal patients, unlike previous studies that included heterogeneous populations of neonates, premenarchal girls, adolescents, and young adults aged up to 20 years [5,6,15]. Our selection enabled us to clearly identify clinical aspects of OT and to help raise the level of awareness of diagnosticians who treat this specific patient population. The lack of specificity of signs and symptoms complicates the diagnosis of OT. Similar to other studies [2,3,7], we did not identify risk factors or symptom(s) that correlated well with an existing OT. Fever or elevated white blood cell count was present in only a few of our patients, and most of them had a normal-sized ovary. Anders et al , Beaunoyer et al , and Muehlstedt et al  also demonstrated normal-sized ovaries in 55%, 49%, and 46% of their cases, respectively. The incidence of OT without any underlying abnormality, however, appears to be more frequent in premenarchal girls compared with adolescents in whom it is usually correlated to the presence of a functional cyst . We did not have any cases of malignant masses, unlike other reports of higher neoplastic cyst rates (benign or malignant) in premenarchal girls . Also, 59% (13/20) of our patients did not have any underlying ovarian disease. Cases of OT of the normal-appearing adnexa seem to occur more frequently in premenarchal girls (15%–50% of cases) . Several theories have been proposed to explain the presence of OT when there is no adnexal enlargement or mass [7,17]. One is excessive mobility of the adnexa caused by an abnormally long oviduct, mesosalpinx, and mesovarium or hyperlaxity of those ligaments; other explanations are attributed to adnexal venous congestion that may follow premenarchal activity or jarring movement of the body [7,17]. 31 The use of AUS scans in the diagnosis of OT was demonstrated to have low specificity . The search for a better modality of imaging in the diagnosis of OT led to the suggestion of Doppler-flow studies used to evaluate compromise in arterial or venous blood flow to the affected ovary. Its role as a predictor of torsion, however, has elicited similar controversy [6,7,17,21]. Servaes et al  demonstrated either arterial or venous blood flow in 64% of their patients. Similarly, Stark and Siegel  noted blood flow in 10 of 15 patients. In contrast, 9 of 10 patients who underwent Doppler imaging by Celik et al  had decreased or absent blood flow, and we demonstrated an absence of flow in all 6 of our patients who underwent Doppler studies. Laparoscopic procedures are superior to open surgery in several aspects: the incisions are smaller, blood loss and analgesic requirements are less, and postoperative recovery is quicker. The advantages of laparoscopy in the diagnosis and treatment of pelvic masses in gynecology are well established [5,14,22,23], and it was the preferred surgical approach in our series as well. Three cases of laparotomy were performed for the following indications: a 3-monthold with signs of peritoneal irritation, a 12-year-old with a large dermoid cyst, and an 11-year-old with an uncertain presurgical diagnosis. In accordance with other reports [6,7,11,24], OT occurred more frequently on the right side in the patients of our study group, with a ratio of about 7:3. This tendency has been attributed to the presence of the sigmoid colon on the left side, which may help to prevent left-sided torsion . Darrell et al  speculated that this difference may result from undiagnosed events of OT on the left side, because the increased suspicion of appendicitis may lead to a more thorough investigation. We are inclined to agree with their line of thought. There has been a growing trend toward a more conservative approach in the treatment of OT over the past decade. Previous reports of successful conservative laparoscopic interventions in premenarchal patients , as well as negligible cases of cancer and data presented below all favor the conservative approach. The advocates for a radical approach (ie, resection of an ischemic-appearing ovary or fallopian tube) raise several arguments against performing detorsion: (1) a black-bluish adnexa signifies an irreversible process of ischemia and necrosis; (2) untwisting of a torsed ovary might dislodge a thrombus/embolus from pelvic veins, causing a thromboembolic event/complication; (3) the concern of malignancy associated with torsed ovaries [7,9]. Mounting evidence in the literature has refuted the first argument, claiming that the gross appearance of a twisted hemorrhagic adnexa does not imply that it is nonviable [15,18,26,27]. For example, Galinier et al  found viable ovarian parenchyma on surgical specimens among 7 of 11 cases in which black-bluish ovaries were detorsed and removed, and Muehlstedt et al  came to similar conclusions. With regard to thrombotic/embolic risks, a thorough review of the literature since 1900 found only 2 cases of 32 pulmonary embolism associated with OT after laparotomy and adnexal resection, and there have been no reported events of thromboembolism after detorsion . The third argument about the possibility of leaving a malignancy in situ, however, is disturbing. The reported malignancy rate for ovarian masses in the pediatric population is 10%, and several series demonstrated that the incidence of malignancy specifically associated with torsion is actually much lower (up to 2%) [5,9–11]. Notably, Sommerville et al  found that benign ovarian neoplasms had a 12.9-fold increased risk of undergoing adnexal torsion compared with malignant ones (p , .001). The authors explained this finding by the tendency of malignant neoplasms to cause inflammation and adhesions to surrounding structures, paradoxically preventing torsion. The presence of an ovarian mass should trigger meticulous clinical assessment to rule out the possibility of malignancy. That in-depth approach includes the exclusion of clinical symptoms and signs of malignancy (mainly menstrual disturbances, pubertal precocity, virilization, ascites, or other signs of metastatic spread) in conjunction with morphologic characterization of the mass by AUS and Doppler flow studies, followed by measurement of tumor markers. These measures ought to reduce the risk of overlooking the presence of a malignancy during a detorsion procedure. Indeed, there has been no report of such a disturbing scenario thus far. Time plays an important role in the effort to conserve the involved adnexa. Rousseau et al  reported an inverse relationship between the time interval from admission to diagnosis and to performing conservative surgery. Contrary to other studies [11,15], conservative treatment was the preferred approach for most of our cases (86.4%). Our ability to safely choose conservative management can be partially attributed to a very short interval between admission to the ER and intraoperative diagnosis and treatment. The average time interval from the onset of pain to surgery in our patients was 54.5 hours (median interval 20.75 hours). It is perhaps this relatively short interval that aided us in the decision to perform conservative surgery, in accordance with the data presented by Rousseau et al . We had 3 cases where radical surgery was performed. Oophorectomy was performed in a 12-year-old with a functional cyst complicated by severe ovarian bleeding; oophorectomy in a 3-month-old admitted 150 hours after onset of pain caused by irreversible necrosis (pathologic report of hemorrhagic necrosis); and partial oophorectomy in a 12-year-old admitted 101 hours after the onset of pain caused by a dermoid cyst. There were 3 cases of recurrent OT in our series, 2 after detorsion alone and 1 after detorsion and cyst drainage. The first recurrence was in an 11-year-old patient 20 months after a detorsion procedure of a normal-appearing ovary. The recurrence was due to a 6-cm cyst that was removed. Oophoropexy was performed by plication of uteroovarian ligament. The second case of recurrence was in a 14-year-old Journal of Minimally Invasive Gynecology, Vol 19, No 1, January/February 2012 patient. The first episode was treated in another hospital and data regarding this event was not available. The recurrence was due to a 6-cm hemorrhagic cyst that was drained. The third recurrence was in a 14-year-old patient 22 months after the first event of torsion of a normal appearing ovary. The recurrence was due to an enlarged ovary without a cyst. Oophoropexy was carried out. The precise risk of recurrent ipsilateral or asynchronous contralateral OT is not known, but it is estimated as being between 2% to 5% [9,18,30,31]. Beaunoyer et al  estimated that the risk of recurrence was 11.4% in OT without ovarian disease. Rousseau et al  reported that 3 of 19 cases treated by detorsion had retorsion, all of which occurred after incomplete removal of the cyst. They concluded that simple puncture or resection of the upper pole of the cysts must be avoided and that the cysts should undergo total resection in order to avoid recurrence. As mentioned earlier, all recurrences in our series occurred after detorsion only or cyst drainage, and none after detorsion combined with cystectomy. The role of oophoropexy in the treatment of children with OT is debatable. Some authors have raised concerns of anatomic alterations decreasing future fertility . There has been no evidence of this sequela in the literature thus far. The general consensus is that the clinical situations that justify the performance of an oophoropexy are postresection of 1 ovary because of torsion, bilateral OT, recurrent OT, and OT without any ovarian disease or a loose or redundant mesosalpinx [7,11,16, 20,33]. Pansky et al  suggested performing oophoropexy in cases of OT in postmenarchal patients with normal appearing ovaries. They describe a relatively high recurrence rate (60%!) if during the primary surgery the adnexa appeared normal . In our series, oophoropexy was performed in 3 cases, 2 after recurrence and 1 after adnexectomy to prevent contralateral OT. Our study has several limitations. It has the drawbacks inherent to any retrospective analysis. Because we located and analyzed only charts of premenarchal girls with OT, we could not deduce rates of occurrence of this condition in young patients presenting with acute abdominal pain. Other nongynecologic cases, as well as cases of OT that we overlooked, were not included in our series. Doppler flow studies were performed in only 6 of our patients. We found a high degree of correlation between abnormal Doppler studies and OT; however, our decision to intervene was clinical rather than based on imaging. Also, Doppler studies were carried out in our ultrasound unit and so were not available during off-hours. The high degree of correlation suggests that further studies should be carried out to define the role of this modality in premenarchal girls. Conclusion A high index of suspicion of OT is cardinal in the evaluation and management of lower abdominal pain in premenarchal patients. The main signs and symptoms of OT are an abrupt onset of lower intermittent abdominal pain and Tsafrir et al. Risk Factors, Symptoms, and Treatment of Ovarian Torsion in Children vomiting. A normal-sized ovary on AUS does not rule out the diagnosis of OT, because a substantial portion of premenarchal patients with OT have no underlying adnexal disease. We contend that the high rate of salvaged ovary (86%) in our current series can be attributable to the short interval between ER admission to diagnosis with the aid of Doppler studies and prompt surgical treatment. A laparoscopic approach to detorsion and conservative surgery should be the treatment of choice. The benefit of ovarian fixation in this age group remains to be established. Acknowledgment We thank Esther Eshkol for editorial assistance. 15. 16. 17. 18. 19. 20. References 21. 1. Guthrie BD, Alder MD, Powell EC. Incidence and trends of pediatric ovarian torsion hospitalizations in the United States, 2000–2006. 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