- Email: [email protected]
Presentation, Diagnosis, and Treatment of Ovarian Torsion in Premenarchal Girls
Accepted Manuscript Presentation, Diagnosis and Treatment of Ovarian Torsion in Premenarchal Girls E. Ashwal, MD, H. Krissi, MD, L. Hiersch, MD, S. Less, MD, R. Eitan, MD, Y. Peled, MD PII:
S1083-3188(15)00166-7
DOI:
10.1016/j.jpag.2015.03.010
Reference:
PEDADO 1839
To appear in:
Journal of Pediatric and Adolescent Gynecology
Received Date: 11 November 2014 Revised Date:
13 March 2015
Accepted Date: 19 March 2015
Please cite this article as: Ashwal E, Krissi H, Hiersch L, Less S, Eitan R, Peled Y, Presentation, Diagnosis and Treatment of Ovarian Torsion in Premenarchal Girls, Journal of Pediatric and Adolescent Gynecology (2015), doi: 10.1016/j.jpag.2015.03.010. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT 1/
ORIGINAL ARTICLE
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Presentation, Diagnosis and Treatment of Ovarian Torsion in Premenarchal Girls
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E. Ashwal MD*, H. Krissi MD*, L. Hiersch MD, S. Less MD, R. Eitan MD,
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Y. Peled MD
Helen Schneider Hospital for Women, Rabin Medical Center, Petach Tikva, and Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel
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*The first two authors contributed equally to the work.
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Correspondence address:
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Yoav Peled, MD Department of Obstetrics and Gynecology Helen Schneider Hospital for Women Rabin Medical Center Petach Tikva 49100, Israel Tel: +972-3-9377400 Fax: +972-3-9377409 E-mail: [email protected]
ACCEPTED MANUSCRIPT 2/ ABSTRACT Study Objective: To describe the clinical characteristics and treatment of ovarian torsion in premenarchal girls with surgically verified ovarian torsion.
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Design and Participants: A retrospective cohort study design was used. The medical charts of all premenarchal girls with surgically verified ovarian torsion
reviewed for clinical, treatment and outcome data.
SC
treated in a university-affiliated tertiary medical center from 1997-2012 were
Results: Thirty-two premenarchal girls were identified. Median age was 9 years.
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There were 7 recurrences altogether during the study period (17.9%) for a total of 39 cases. The main presenting symptoms were abdominal pain (92.3%) and nausea and vomiting (84.6%). Physical examination revealed abdominal tenderness in 25 cases (64.1%). Abdominal ultrasound, performed in 31 patients
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(38 cases), yielded pathological findings in 28 (73.7%), mainly an enlarged ovary (11 cases, 28.9%). Doppler flow studies were abnormal in 15 cases. In 26 cases (68.4%), the tentative preoperative working diagnosis was ovarian torsion.
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Laparoscopy was performed in 26 cases, laparotomy in 10 and laparoscopy converted to laparotomy in 3 cases. Conservative management, mainly detorsion
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with additional cyst drainage or cystectomy, was used in 37 cases (95.2%) with oophoropexy in 5 cases. Two patients required oophorectomy because of a suspected neoplasm and severe ovarian necrosis. Pathologic examination demonstrated 5 simple cysts, one necrotic ovary and one mature cystic teratoma. Conclusions: Ovarian torsion in premenarchal girls is associated with nonspecific signs and symptoms. Abdominal ultrasound and Doppler imaging may assist in
ACCEPTED MANUSCRIPT 3/ the diagnosis. Laparoscopy with conservative management is preferred. Owing
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to the high recurrence rate, oophoropexy may be considered.
Key words: Ovarian torsion, Premenarch, Adnexal torsion, Conservative
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surgery, Ultrasound
ACCEPTED MANUSCRIPT 4/ Introduction Torsion of the ovary, tube or both is considered to account for 2.7% of all gynecologic emergencies.(1) It occurs mostly in patients of child-bearing age (2)
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and is less common in the 1-20-year age group in which the reported prevalence is 4.9 per 100,000 females aged 1 to 20 years.(3) Signs and symptoms are nonspecific and even with the help of laboratory tests, sonography and Doppler-
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flow studies, ovarian torsion is frequently misdiagnosed.(2, 4, 5) This leads to a delay in diagnosis and a long lag time of hours to days from symptom onset to
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surgery.(2) An interval of more than 10 hours is associated with adnexal necrosis, but the actual duration of ischemia beyond which the damage is irreversible remains unknown.
The preferred treatment of confirmed ovarian torsion is controversial. A
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growing number of studies advocate conservative surgery, mainly detorsion with additional cyst drainage or cystectomy; however, all have so far been limited to adult patients.(7-11) Although resection of the affected adnexa can have an
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adverse impact on future reproductive potential, (6) concerns about missing a malignant lesion, thromboembolic complications and a severe ischemic and
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nonviable ovary have led to the use of oophorectomy. (7-11) A few recent reports have focused on ovarian torsion in the pediatric
population (5, 8, 12) but almost none stratified for premenarchal girls. Therefore, the aim of the present study was to characterize the signs, symptoms and treatment of ovarian torsion in premenarchal girls.
ACCEPTED MANUSCRIPT 5/ Methods Study Population and Setting The study cohort consisted of all premenarchal patients with surgically
tertiary medical center from 1997 to 2012.
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verified ovarian torsion who were diagnosed and treated in a university-affiliated
According to our local departmental protocols, every girl with abdominal
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pain admitted to the emergency room, either pediatric or gynecologic, undergoes a meticulous history, physical examination and urine analysis. Further laboratory
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tests (complete blood count, electrolytes, liver and kidney function tests) are performed at the discretion of the attending physician. In addition, abdominal ultrasound is performed with a full bladder, including pelvic sonography with Doppler flow imaging. When ovarian/adnexal torsion is suspected, surgery is
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performed by gynecologic personnel. The surgical approach is usually based on the surgeon’s preference.
For the present study, cases of ovarian/adnexal torsion were identified
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retrospectively using the hospital’s comprehensive computerized database. Eligibility was based on ICD-9 codes for ovarian/adnexal torsion. Data were
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collected from the patient files and electronic records as follows: patient age; medical and gynecologic history; presenting symptoms and signs; findings on physical examination, laboratory work-up and imaging studies; time elapsed from symptom onset to emergency room (either pediatric or gynecologic) admission, from emergency room admission to gynecologic evaluation and from emergency room admission to first incision made in the operating room; surgical findings;
ACCEPTED MANUSCRIPT 6/ surgical treatment of the ovarian torsion; and postoperative complications. We also recorded whether the index ovarian torsion was a primary event or a recurrence. In cases in which the ovarian vessels were wrapped around a central
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axis in a clockwise or counterclockwise direction, a whirlpool sign on Doppler imaging was considered.
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The study protocol was approved by the local Institutional Review Board.
Data Analysis
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Data analysis was performed with the SPSS package, v19.0 (Chicago, IL). Normally distributed variable are reported as means and standard deviations and non-normally distributed variables as medians.
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Results Background Characteristics
A total of 32 premenarchal patients met the study criteria. Median age was
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9 years (range 3 months to 14 years); mean age was 7.7 years and standard deviation was 4.4 years. During the study period, ovarian torsion recurred in 6
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patients (18.7%), for a total of 39 cases: 5 patients had two episodes of ovarian torsion each and one patient had three episodes. Previous abdominal surgery was performed in one patient and another 6 patients had a history of pelvic surgeries.
ACCEPTED MANUSCRIPT 8/ Physical Examination (Table 1) The median interval from symptom onset to emergency room admission was 24 hours. The most common presenting symptom was abdominal pain
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(36/39 episodes of torsion, 92.3%), followed by nausea and/or vomiting (33/39, 84.6%). Other gastrointestinal and urinary complaints are listed in Table 1. The most common sign on physical examination was abdominal tenderness in 25
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cases (64.1%), followed by high fever (>38.0°C) in 5 cases (12.8%). Leukocyte
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level was elevated (>12 X 10³/µL) in 15 cases (38.4%).
Ultrasound and Doppler Imaging (Table 2)
Abdominal ultrasound with a full bladder, performed in 31 patients (38 cases, 97.4%), revealed a pathologic pelvic finding in 28 cases (73.7%), most
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commonly an enlarged ovary (≥4cm in at least in one dimension) (11 cases, 28.9%) and ovary edema (9 cases, 23.6%); defined as, an accumulation of fluid within the ovarian stroma separating normal follicular structures. Doppler flow
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studies were considered pathological in 15 cases and, additionally, a whirlpool sign was observed in 2 cases. Additional findings included simple and complex
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cysts. Ovarian torsion was suspected by abdominal ultrasound in 26 cases (68.4%). Computed tomography was performed in only 2 patients and demonstrated an ovarian mass.
Surgical Findings and Treatment (Table 3)
ACCEPTED MANUSCRIPT 9/ The median interval from emergency room admission to the first incision in the operating room was 9.5 hours. The majority of cases of torsion occurred on the left side (53.5%). Laparoscopy was performed in 26 cases and laparotomy in
surgery was primarily based on surgeon preference.
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10; in the remaining 3 cases, laparoscopy was converted to laparotomy. Mode of
The median number of adnexal twists was 2.5 (range 1–5). Additional
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pathologic findings at surgery included a bluish-black ovary in 25 cases, an enlarged ovary in 13 and ovarian or para-ovarian cysts in 10 cases (7 simple, 3
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complex according to the pathologic report). Other concomitant findings were hydrosalpinx, inguinal hernia and appendicitis.
Conservative management, mainly detorsion with either additional cyst drainage or cystectomy, was the standard of care in 37 cases (94.9%). In 5
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cases, oophoropexy consisting of plication of the ovarian ligament was performed as well. The remaining 2 cases were treated by oophorectomy because of a suspected neoplasm on macroscopic examination (mature cystic
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teratoma in the pathology report) in one case and severe ovarian necrosis in the
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other. No malignant neoplasms were identified in any of our patients.
Discussion
The present study describes the symptoms, signs and treatment of
premenarchal ovarian torsion. Unlike previous studies (5, 12), our study population was restricted to a homogenous group of premenarchal girls all attending the same university-affiliated tertiary medical center.
ACCEPTED MANUSCRIPT 10/ Ovarian torsion poses a diagnostic challenge because clinicians often have difficulty differentiating it from other conditions involving the lower abdomen. In our study, the median interval from symptom onset to emergency room
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admission was 24 hours and from emergency room admission to surgery, 9.5 hours. Delays in diagnosis have also been reported by others, ranging from several hours(13) and days, (14) as in the present study, to months (15, 16) and
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even years. The majority of our patients reported acute onset of abdominal pain along with nausea and vomiting. Only a few presented with pyrexia or elevated
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white blood cell count. Similar findings were reported by others. (17, 18) Normal-sized ovaries were noted on abdominal ultrasound in 71.0% of our cases (28/38). This rate is higher than the 45-51% reported in studies of ovarian torsion in adolescent girls or women (4, 19) but close to the 60% rate reported by
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Tsafrir et al. (24) in premenarchal girls. These findings suggest that ovarian torsion with normal-appearing ovaries is more characteristic of the younger age group. Several researchers suggested that in these cases, the ovarian torsion
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may be attributable to the presence of an abnormally long tube, mesosalpinx, mesovarium or adnexal venous congestion due to premenstrual hormone activity
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or jarring movements of the body. (20, 21) In 16.2% of cases (10/38), normal sonographic appearance of the adnexa was noted, reflecting the inherent difficulty of performing and interpreting sonographic evaluation in this specific population.
We used Doppler-flow studies to determine if arterial or venous blood flow to the affected adnexa was compromised. The results were abnormal in only 17
ACCEPTED MANUSCRIPT 11/ cases (44.7%). However, the predictive role of Doppler imaging in ovarian torsion is still unclear, with some authors reporting arterial or venous blood flow
much higher rates of 90-100%.(18, 21)
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abnormalities in approximately two-thirds of patients (12, 22) and others noting
Ovarian torsion in our cohort occurred slightly more often on the left side (53.8%), contrary to previous reports in adolescents and women.(13, 18, 23, 24) torsion in older patients may be explained by the
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The lower rate of left
decreased mobility of the left adnexa due to its partial adhesion to the
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mesosigmoid (13) or to an increased suspicion of appendicitis leading to a relatively higher rate of right-side diagnoses.(24) Neither of these factors seems to be relevant to the premenarchal population.
In the present study, a bluish-black ovary was observed during
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laparoscopy/laparotomy in 25 cases, reflecting the delay in diagnosis. Nevertheless, in all cases except 2, the adnexa was conserved. There is mounting evidence that the gross appearance of a twisted hemorrhagic adnexa
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may be misleading, as the ovary retains viability despite assumable prolonged ischemia in 88% to 100% of cases. (8, 25) This suggests that complete arterial
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obstruction does not occur. Therefore, we believe, oophorectomy should be avoided. As only a minimal blood supply is derived from the ovarian or uterine arteries, the edema and enlargement of the ovary is caused by lymphatic stasis and the hemorrhagic appearance is caused by blue-black appearance rather than actual gangrene.(26)
ACCEPTED MANUSCRIPT 12/ No malignant neoplasms were identified in any of our patients. A benign neoplasm or cyst accounted for only 16 cases of torsion (41.0%). Similarly, previous studies found that 97% of cases of adnexal torsion were associated with
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normal ovaries or ovaries with benign pathology, (16) and a low risk of malignancy and thromboembolism.(16,26,30) The presence of malignant lesions caused more inflammation and fibrosis, leading to adherence of the ovary to
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surrounding structures.(13) Thus, adnexal conservation surgery performed as
torsion in childhood.(14)
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soon as possible seems to be the best surgical approach, especially in cases of
The association of symptom duration with severity of the ovarian infarction is still unclear. Some authors reported no direct correlation (6) whereas others noted an inverse relationship between the time from admission and performance
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of conservative surgery (adnexa/ovarian sparing surgery). In the present study, due to the relatively shorter interval from emergency room admission to treatment (median 9.5 hours), conservative surgery could be applied in 94.8% of
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cases, contrary to previous studies.(23)
There were 7 cases of recurrent ovarian torsions in 6 patients in our
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series, for a rate of 18.7%. Six occurred in patents with normal-appearing ovaries who previously underwent detorsion alone. (In one patient, the first episode was treated in a different hospital and data were unavailable). The risk of recurrent ipsilateral or asynchronous contralateral ovarian torsion is unknown, though estimates range from 2% to 5% (26, 27) to as high as 10% to 11.4% in the absence of apparent ovarian disease (18, 19). Previous studies suggested that
ACCEPTED MANUSCRIPT 13/ simple puncture or resection of only the upper pole of the cysts is not sufficient and cysts should be completely resected in order to avoid recurrence. (18, 23) Our findings raise the possibility that fixation should be performed in cases of
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adnexal/ovary torsion in premenarchal patients with normal-appearing adnexa. A similar suggestion was made by Pansky et al. (31) in a study of postmenarchal ovarian torsion.
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Our study is one of the largest retrospective studies to date on ovarian torsion in premenarchal girls. However, since we analyzed only medical charts of
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premenarchal girls with surgically proven ovarian torsion, any rates of postmenarchal recurrence could not be deduced. Additionally, due to its retrospective nature of our study, we were not able to correctly distinguish between patients that were prepubertal and those who were merely
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premenarchal at admission.
ACCEPTED MANUSCRIPT 14/ Conclusion Ovarian torsion is a surgical emergency. Owing to its potentially grave consequences and relatively high rate of recurrence, its presence should be
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considered in all premenarchal patients who present with acute abdominal pain. Laparoscopy with conservative surgery (detorsion and/or cystectomy) is the
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treatment of choice.
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ACCEPTED MANUSCRIPT References 1.
Hibbard LT: Adnexal torsion. Am J Obstet Gynecol 1985; 152:456
2.
Houry D, Abbott JT: Ovarian torsion: a fifteen-year review. Ann Emerg Med 2001; 38:156 Guthrie BD, Adler MD, Powell EC: Incidence and trends of pediatric
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3.
ovarian torsion hospitalizations in the United States, 2000-2006. Pediatrics 2010; 125:532
Mazouni C, Bretelle F, Menard JP, et al: [Diagnosis of adnexal torsion
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4.
and predictive factors of adnexal necrosis]. [Article in French] Gynecol
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Obstet Fertil 2005; 33:102 5.
Oltmann SC, Fischer A, Barber R, et al: Cannot exclude torsion -- a 15year review. J Pediatr Surg 2009; 44:1212
6.
Anders JF, Powell EC: Urgency of evaluation and outcome of acute
159:532
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ovarian torsion in pediatric patients. Arch Pediatr Adolesc Med 2005;
Cass DL: Ovarian torsion. Seinm Pediatr Surg 2005; 14:86
8.
Cohen Z, Shinhar D, Kopernik G, et al: The laparoscopic approach to
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7.
9.
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uterine adnexal torsion in childhood. J Pediatr Surg 1996; 31:1557 Aziz D, Davis V, Allen L, et al: Ovarian torsion in children: is oophorectomy necessary? J Pediatr Surg 2004; 39:750
10.
Rousseau V, Massicot R, Darwish AA, et al: Emergency management and conservative surgery of ovarian torsion in children: a report of 40 cases. J Pediatr Aadolesc Gynecol 2008; 21:201
11.
Shalev J, Goldenberg M, Oelsner G, et al: Treatment of twisted ischemic adnexa by simple detorsion. New Engl J Med 1989; 321:546
16/
ACCEPTED MANUSCRIPT 12.
Varras M, Akrivis C, Demou A, et al: Asynchronous bilateral adnexal torsion in a 13-year-old adolescent: our experience of a rare case with review of the literature. J Adolesc Health 2005; 37:244
13.
Templeman C, Hertweck SP, Fallat ME: The clinical course of
14.
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unresected ovarian torsion. J Pediatr Surg 2000; 35:1385
Galinier P, Carfagna L, Delsol M, et al: Ovarian torsion. Management and ovarian prognosis: a report of 45 cases. J Pediatr Surg 2009;
15.
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44:1759
Savic D, Stankovic ZB, Djukic M, et al: Torsion of malignant ovarian
2008; 21:1073 16.
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tumors in childhood and adolescence. J Pediatr Endocrinol Metab
Gocmen A, Karaca M, Sari A: Conservative laparoscopic approach to adnexal torsion. Arch Gynecol Obstet 2008; 277:535 Servaes S, Zurakowski D, Laufer MR, et al: Sonographic findings of
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17.
ovarian torsion in children. Pediatr Radiol 2007; 37:446 18.
Kokoska ER, Keller MS, Weber TR: Acute ovarian torsion in children.
Ozcan C, Celik A, Ozok G, et al: Adnexal torsion in children may have
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19.
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Am J Surg 2000; 180:462
a catastrophic sequel: asynchronous bilateral torsion. J Pediatr Surg 2002; 37:1617
20.
Breech LL, Hillard PJ. Adnexal torsion in pediatric and adolescent girls. Curr Opin Obstet Gynecol 2005; 17:483
21.
Descargues G, Tinlot-Mauger F, Gravier A, et al: Adnexal torsion: a report on forty-five cases. Eur J Obstet Gynecol Reprod Biol 2001; 98:91
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ACCEPTED MANUSCRIPT 22.
Beaunoyer M, Chapdelaine J, Bouchard S, et al: Asynchronous bilateral ovarian torsion. J Pediatr Surg 2004; 39:746
23.
Oelsner G, Shashar D: Adnexal torsion. Clin Obstet Gynecol 2006; 49:459 Tsafrir Z, Azem F, Hasson J, et al: Risk factors, symptoms and
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24.
treatment of ovarian torsion in children: the twelve-year experience of one center. J Minim Invasive Gynecol 2012; 19:29
McCrea RS: Uterine adnexal torsion with subsequent contralateral recurrence. J Reprod Med 1980; 25:123
Celik A, Ergun O, Aldemir H, et al: Long-term results of conservative
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26.
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25.
management of adnexal torsion in children. J Pediatr Surg 2005; 40:704 27.
Stark JE, Siegel MJ: Ovarian torsion in prepubertal and pubertal girls:
28.
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sonographic findings. AJR Am J Roentgenol 1994; 163:1479 Kruger E, Heller DS: Adnexal torsion. A clinicopathologic review of 31 cases. J Reprod Med 1999; 44:71 Oelsner G, Cohen SB, Soriano D, et al: Minimal surgery for the twisted
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29.
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ischaemic adnexa can preserve ovarian function. Hum Reprod 2003; 18:2599
30.
Oltmann SC, Fischer A, Barber R, et al: Pediatric ovarian malignancy presenting as ovarian torsion: incidence and relevance. J Pediatr Surg 2010; 45:135
31.
Pansky M, Smorgick N, Herman A, et al: Torsion of normal adnexa in postmenarchal women and risk of recurrence. Obstet Gynecol 2007; 109:355
ACCEPTED MANUSCRIPT Table 1 Signs and symptoms of ovarian torsion in 32
N (%)
Abdominal pain
36 (92.3)
Lower abdomen
24 (61.5)
Diffuse
12 (30.7) 33 (84.6)
Diarrhea
3 (7.6)
Restlessness
5 (12.8)
Urinary symptoms
3 (7.6)
Fever >38°C
5 (12.8)
Abdominal tenderness Abdominal distension Pelvic mass
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Nausea and vomiting
25 (64.1) 4 (10.2) 3 (7.6)
15 (38.4)
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White blood cells >12x10³/µL
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Signs and Symptoms
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premenarchal patients (39 cases)
ACCEPTED MANUSCRIPT Table 2 Abdominal ultrasound findings associated with ovarian torsion in 31 premenarchal patients (38 cases) N (%)
Any Abnormal finding
28 (73.7)
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Ultrasound Findings
11 (28.9)
Edema of the ovary
9 (23.6)
Enhanced echogenicity
1 (2.6)
Free pelvic fluid
3 (7.9)
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Enlarged ovary
Abnormal Doppler Flow
1 (2.6)
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Absence of venous flow Absence of arterial and venous blood flow
14 (36.8)
Whirlpool sign
2 (5.2)
Additional Findings Simple cyst
11 (28.9)
Complex cyst
4 (10.5)
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Normal abdominal ultrasound
10 (26.3)
ACCEPTED MANUSCRIPT Table 3 Additional pathologic findings at surgery and surgical treatment in 32 premenarchal patients with ovarian torsion
Findings and Procedure
N (%)
Affected Side 18 (46.1)
Left
21 (53.8)
Additional Pathologic Findings
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Right
25 (64.1)
Enlarged ovary (>4cm)
13 (33.3)
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Black-bluish ovary Simple ovarian/para-ovarian cyst
7 (17.9)
Complex ovarian/para-ovarian cyst
3 (7.6)
Hemorrhagic cyst
1 (2.5)
Hydrosalpinx
1 (2.5)
Appendicitis
3 (7.6)
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Inguinal hernia
3 (7.6)
Surgical Procedure Detorsion
20 (51.2) 9 (23.0)
Detorsion and cystectomy
5 (12.8)
Detorsion and fixation
6 (15.3)
Ovarian resection
2 (5.1)
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Detorsion and drainage
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(39 cases)
S1083-3188(15)00166-7
DOI:
10.1016/j.jpag.2015.03.010
Reference:
PEDADO 1839
To appear in:
Journal of Pediatric and Adolescent Gynecology
Received Date: 11 November 2014 Revised Date:
13 March 2015
Accepted Date: 19 March 2015
Please cite this article as: Ashwal E, Krissi H, Hiersch L, Less S, Eitan R, Peled Y, Presentation, Diagnosis and Treatment of Ovarian Torsion in Premenarchal Girls, Journal of Pediatric and Adolescent Gynecology (2015), doi: 10.1016/j.jpag.2015.03.010. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT 1/
ORIGINAL ARTICLE
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Presentation, Diagnosis and Treatment of Ovarian Torsion in Premenarchal Girls
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E. Ashwal MD*, H. Krissi MD*, L. Hiersch MD, S. Less MD, R. Eitan MD,
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Y. Peled MD
Helen Schneider Hospital for Women, Rabin Medical Center, Petach Tikva, and Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel
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*The first two authors contributed equally to the work.
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Correspondence address:
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Yoav Peled, MD Department of Obstetrics and Gynecology Helen Schneider Hospital for Women Rabin Medical Center Petach Tikva 49100, Israel Tel: +972-3-9377400 Fax: +972-3-9377409 E-mail: [email protected]
ACCEPTED MANUSCRIPT 2/ ABSTRACT Study Objective: To describe the clinical characteristics and treatment of ovarian torsion in premenarchal girls with surgically verified ovarian torsion.
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Design and Participants: A retrospective cohort study design was used. The medical charts of all premenarchal girls with surgically verified ovarian torsion
reviewed for clinical, treatment and outcome data.
SC
treated in a university-affiliated tertiary medical center from 1997-2012 were
Results: Thirty-two premenarchal girls were identified. Median age was 9 years.
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There were 7 recurrences altogether during the study period (17.9%) for a total of 39 cases. The main presenting symptoms were abdominal pain (92.3%) and nausea and vomiting (84.6%). Physical examination revealed abdominal tenderness in 25 cases (64.1%). Abdominal ultrasound, performed in 31 patients
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(38 cases), yielded pathological findings in 28 (73.7%), mainly an enlarged ovary (11 cases, 28.9%). Doppler flow studies were abnormal in 15 cases. In 26 cases (68.4%), the tentative preoperative working diagnosis was ovarian torsion.
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Laparoscopy was performed in 26 cases, laparotomy in 10 and laparoscopy converted to laparotomy in 3 cases. Conservative management, mainly detorsion
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with additional cyst drainage or cystectomy, was used in 37 cases (95.2%) with oophoropexy in 5 cases. Two patients required oophorectomy because of a suspected neoplasm and severe ovarian necrosis. Pathologic examination demonstrated 5 simple cysts, one necrotic ovary and one mature cystic teratoma. Conclusions: Ovarian torsion in premenarchal girls is associated with nonspecific signs and symptoms. Abdominal ultrasound and Doppler imaging may assist in
ACCEPTED MANUSCRIPT 3/ the diagnosis. Laparoscopy with conservative management is preferred. Owing
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to the high recurrence rate, oophoropexy may be considered.
Key words: Ovarian torsion, Premenarch, Adnexal torsion, Conservative
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surgery, Ultrasound
ACCEPTED MANUSCRIPT 4/ Introduction Torsion of the ovary, tube or both is considered to account for 2.7% of all gynecologic emergencies.(1) It occurs mostly in patients of child-bearing age (2)
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and is less common in the 1-20-year age group in which the reported prevalence is 4.9 per 100,000 females aged 1 to 20 years.(3) Signs and symptoms are nonspecific and even with the help of laboratory tests, sonography and Doppler-
SC
flow studies, ovarian torsion is frequently misdiagnosed.(2, 4, 5) This leads to a delay in diagnosis and a long lag time of hours to days from symptom onset to
M AN U
surgery.(2) An interval of more than 10 hours is associated with adnexal necrosis, but the actual duration of ischemia beyond which the damage is irreversible remains unknown.
The preferred treatment of confirmed ovarian torsion is controversial. A
TE D
growing number of studies advocate conservative surgery, mainly detorsion with additional cyst drainage or cystectomy; however, all have so far been limited to adult patients.(7-11) Although resection of the affected adnexa can have an
EP
adverse impact on future reproductive potential, (6) concerns about missing a malignant lesion, thromboembolic complications and a severe ischemic and
AC C
nonviable ovary have led to the use of oophorectomy. (7-11) A few recent reports have focused on ovarian torsion in the pediatric
population (5, 8, 12) but almost none stratified for premenarchal girls. Therefore, the aim of the present study was to characterize the signs, symptoms and treatment of ovarian torsion in premenarchal girls.
ACCEPTED MANUSCRIPT 5/ Methods Study Population and Setting The study cohort consisted of all premenarchal patients with surgically
tertiary medical center from 1997 to 2012.
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verified ovarian torsion who were diagnosed and treated in a university-affiliated
According to our local departmental protocols, every girl with abdominal
SC
pain admitted to the emergency room, either pediatric or gynecologic, undergoes a meticulous history, physical examination and urine analysis. Further laboratory
M AN U
tests (complete blood count, electrolytes, liver and kidney function tests) are performed at the discretion of the attending physician. In addition, abdominal ultrasound is performed with a full bladder, including pelvic sonography with Doppler flow imaging. When ovarian/adnexal torsion is suspected, surgery is
TE D
performed by gynecologic personnel. The surgical approach is usually based on the surgeon’s preference.
For the present study, cases of ovarian/adnexal torsion were identified
EP
retrospectively using the hospital’s comprehensive computerized database. Eligibility was based on ICD-9 codes for ovarian/adnexal torsion. Data were
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collected from the patient files and electronic records as follows: patient age; medical and gynecologic history; presenting symptoms and signs; findings on physical examination, laboratory work-up and imaging studies; time elapsed from symptom onset to emergency room (either pediatric or gynecologic) admission, from emergency room admission to gynecologic evaluation and from emergency room admission to first incision made in the operating room; surgical findings;
ACCEPTED MANUSCRIPT 6/ surgical treatment of the ovarian torsion; and postoperative complications. We also recorded whether the index ovarian torsion was a primary event or a recurrence. In cases in which the ovarian vessels were wrapped around a central
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axis in a clockwise or counterclockwise direction, a whirlpool sign on Doppler imaging was considered.
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The study protocol was approved by the local Institutional Review Board.
Data Analysis
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Data analysis was performed with the SPSS package, v19.0 (Chicago, IL). Normally distributed variable are reported as means and standard deviations and non-normally distributed variables as medians.
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Results Background Characteristics
A total of 32 premenarchal patients met the study criteria. Median age was
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9 years (range 3 months to 14 years); mean age was 7.7 years and standard deviation was 4.4 years. During the study period, ovarian torsion recurred in 6
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patients (18.7%), for a total of 39 cases: 5 patients had two episodes of ovarian torsion each and one patient had three episodes. Previous abdominal surgery was performed in one patient and another 6 patients had a history of pelvic surgeries.
ACCEPTED MANUSCRIPT 8/ Physical Examination (Table 1) The median interval from symptom onset to emergency room admission was 24 hours. The most common presenting symptom was abdominal pain
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(36/39 episodes of torsion, 92.3%), followed by nausea and/or vomiting (33/39, 84.6%). Other gastrointestinal and urinary complaints are listed in Table 1. The most common sign on physical examination was abdominal tenderness in 25
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cases (64.1%), followed by high fever (>38.0°C) in 5 cases (12.8%). Leukocyte
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level was elevated (>12 X 10³/µL) in 15 cases (38.4%).
Ultrasound and Doppler Imaging (Table 2)
Abdominal ultrasound with a full bladder, performed in 31 patients (38 cases, 97.4%), revealed a pathologic pelvic finding in 28 cases (73.7%), most
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commonly an enlarged ovary (≥4cm in at least in one dimension) (11 cases, 28.9%) and ovary edema (9 cases, 23.6%); defined as, an accumulation of fluid within the ovarian stroma separating normal follicular structures. Doppler flow
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studies were considered pathological in 15 cases and, additionally, a whirlpool sign was observed in 2 cases. Additional findings included simple and complex
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cysts. Ovarian torsion was suspected by abdominal ultrasound in 26 cases (68.4%). Computed tomography was performed in only 2 patients and demonstrated an ovarian mass.
Surgical Findings and Treatment (Table 3)
ACCEPTED MANUSCRIPT 9/ The median interval from emergency room admission to the first incision in the operating room was 9.5 hours. The majority of cases of torsion occurred on the left side (53.5%). Laparoscopy was performed in 26 cases and laparotomy in
surgery was primarily based on surgeon preference.
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10; in the remaining 3 cases, laparoscopy was converted to laparotomy. Mode of
The median number of adnexal twists was 2.5 (range 1–5). Additional
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pathologic findings at surgery included a bluish-black ovary in 25 cases, an enlarged ovary in 13 and ovarian or para-ovarian cysts in 10 cases (7 simple, 3
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complex according to the pathologic report). Other concomitant findings were hydrosalpinx, inguinal hernia and appendicitis.
Conservative management, mainly detorsion with either additional cyst drainage or cystectomy, was the standard of care in 37 cases (94.9%). In 5
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cases, oophoropexy consisting of plication of the ovarian ligament was performed as well. The remaining 2 cases were treated by oophorectomy because of a suspected neoplasm on macroscopic examination (mature cystic
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teratoma in the pathology report) in one case and severe ovarian necrosis in the
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other. No malignant neoplasms were identified in any of our patients.
Discussion
The present study describes the symptoms, signs and treatment of
premenarchal ovarian torsion. Unlike previous studies (5, 12), our study population was restricted to a homogenous group of premenarchal girls all attending the same university-affiliated tertiary medical center.
ACCEPTED MANUSCRIPT 10/ Ovarian torsion poses a diagnostic challenge because clinicians often have difficulty differentiating it from other conditions involving the lower abdomen. In our study, the median interval from symptom onset to emergency room
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admission was 24 hours and from emergency room admission to surgery, 9.5 hours. Delays in diagnosis have also been reported by others, ranging from several hours(13) and days, (14) as in the present study, to months (15, 16) and
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even years. The majority of our patients reported acute onset of abdominal pain along with nausea and vomiting. Only a few presented with pyrexia or elevated
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white blood cell count. Similar findings were reported by others. (17, 18) Normal-sized ovaries were noted on abdominal ultrasound in 71.0% of our cases (28/38). This rate is higher than the 45-51% reported in studies of ovarian torsion in adolescent girls or women (4, 19) but close to the 60% rate reported by
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Tsafrir et al. (24) in premenarchal girls. These findings suggest that ovarian torsion with normal-appearing ovaries is more characteristic of the younger age group. Several researchers suggested that in these cases, the ovarian torsion
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may be attributable to the presence of an abnormally long tube, mesosalpinx, mesovarium or adnexal venous congestion due to premenstrual hormone activity
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or jarring movements of the body. (20, 21) In 16.2% of cases (10/38), normal sonographic appearance of the adnexa was noted, reflecting the inherent difficulty of performing and interpreting sonographic evaluation in this specific population.
We used Doppler-flow studies to determine if arterial or venous blood flow to the affected adnexa was compromised. The results were abnormal in only 17
ACCEPTED MANUSCRIPT 11/ cases (44.7%). However, the predictive role of Doppler imaging in ovarian torsion is still unclear, with some authors reporting arterial or venous blood flow
much higher rates of 90-100%.(18, 21)
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abnormalities in approximately two-thirds of patients (12, 22) and others noting
Ovarian torsion in our cohort occurred slightly more often on the left side (53.8%), contrary to previous reports in adolescents and women.(13, 18, 23, 24) torsion in older patients may be explained by the
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The lower rate of left
decreased mobility of the left adnexa due to its partial adhesion to the
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mesosigmoid (13) or to an increased suspicion of appendicitis leading to a relatively higher rate of right-side diagnoses.(24) Neither of these factors seems to be relevant to the premenarchal population.
In the present study, a bluish-black ovary was observed during
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laparoscopy/laparotomy in 25 cases, reflecting the delay in diagnosis. Nevertheless, in all cases except 2, the adnexa was conserved. There is mounting evidence that the gross appearance of a twisted hemorrhagic adnexa
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may be misleading, as the ovary retains viability despite assumable prolonged ischemia in 88% to 100% of cases. (8, 25) This suggests that complete arterial
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obstruction does not occur. Therefore, we believe, oophorectomy should be avoided. As only a minimal blood supply is derived from the ovarian or uterine arteries, the edema and enlargement of the ovary is caused by lymphatic stasis and the hemorrhagic appearance is caused by blue-black appearance rather than actual gangrene.(26)
ACCEPTED MANUSCRIPT 12/ No malignant neoplasms were identified in any of our patients. A benign neoplasm or cyst accounted for only 16 cases of torsion (41.0%). Similarly, previous studies found that 97% of cases of adnexal torsion were associated with
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normal ovaries or ovaries with benign pathology, (16) and a low risk of malignancy and thromboembolism.(16,26,30) The presence of malignant lesions caused more inflammation and fibrosis, leading to adherence of the ovary to
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surrounding structures.(13) Thus, adnexal conservation surgery performed as
torsion in childhood.(14)
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soon as possible seems to be the best surgical approach, especially in cases of
The association of symptom duration with severity of the ovarian infarction is still unclear. Some authors reported no direct correlation (6) whereas others noted an inverse relationship between the time from admission and performance
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of conservative surgery (adnexa/ovarian sparing surgery). In the present study, due to the relatively shorter interval from emergency room admission to treatment (median 9.5 hours), conservative surgery could be applied in 94.8% of
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cases, contrary to previous studies.(23)
There were 7 cases of recurrent ovarian torsions in 6 patients in our
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series, for a rate of 18.7%. Six occurred in patents with normal-appearing ovaries who previously underwent detorsion alone. (In one patient, the first episode was treated in a different hospital and data were unavailable). The risk of recurrent ipsilateral or asynchronous contralateral ovarian torsion is unknown, though estimates range from 2% to 5% (26, 27) to as high as 10% to 11.4% in the absence of apparent ovarian disease (18, 19). Previous studies suggested that
ACCEPTED MANUSCRIPT 13/ simple puncture or resection of only the upper pole of the cysts is not sufficient and cysts should be completely resected in order to avoid recurrence. (18, 23) Our findings raise the possibility that fixation should be performed in cases of
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adnexal/ovary torsion in premenarchal patients with normal-appearing adnexa. A similar suggestion was made by Pansky et al. (31) in a study of postmenarchal ovarian torsion.
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Our study is one of the largest retrospective studies to date on ovarian torsion in premenarchal girls. However, since we analyzed only medical charts of
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premenarchal girls with surgically proven ovarian torsion, any rates of postmenarchal recurrence could not be deduced. Additionally, due to its retrospective nature of our study, we were not able to correctly distinguish between patients that were prepubertal and those who were merely
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premenarchal at admission.
ACCEPTED MANUSCRIPT 14/ Conclusion Ovarian torsion is a surgical emergency. Owing to its potentially grave consequences and relatively high rate of recurrence, its presence should be
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considered in all premenarchal patients who present with acute abdominal pain. Laparoscopy with conservative surgery (detorsion and/or cystectomy) is the
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treatment of choice.
15/
ACCEPTED MANUSCRIPT References 1.
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Houry D, Abbott JT: Ovarian torsion: a fifteen-year review. Ann Emerg Med 2001; 38:156 Guthrie BD, Adler MD, Powell EC: Incidence and trends of pediatric
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Shalev J, Goldenberg M, Oelsner G, et al: Treatment of twisted ischemic adnexa by simple detorsion. New Engl J Med 1989; 321:546
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ACCEPTED MANUSCRIPT 12.
Varras M, Akrivis C, Demou A, et al: Asynchronous bilateral adnexal torsion in a 13-year-old adolescent: our experience of a rare case with review of the literature. J Adolesc Health 2005; 37:244
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Descargues G, Tinlot-Mauger F, Gravier A, et al: Adnexal torsion: a report on forty-five cases. Eur J Obstet Gynecol Reprod Biol 2001; 98:91
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ACCEPTED MANUSCRIPT 22.
Beaunoyer M, Chapdelaine J, Bouchard S, et al: Asynchronous bilateral ovarian torsion. J Pediatr Surg 2004; 39:746
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Oelsner G, Shashar D: Adnexal torsion. Clin Obstet Gynecol 2006; 49:459 Tsafrir Z, Azem F, Hasson J, et al: Risk factors, symptoms and
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ACCEPTED MANUSCRIPT Table 1 Signs and symptoms of ovarian torsion in 32
N (%)
Abdominal pain
36 (92.3)
Lower abdomen
24 (61.5)
Diffuse
12 (30.7) 33 (84.6)
Diarrhea
3 (7.6)
Restlessness
5 (12.8)
Urinary symptoms
3 (7.6)
Fever >38°C
5 (12.8)
Abdominal tenderness Abdominal distension Pelvic mass
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Nausea and vomiting
25 (64.1) 4 (10.2) 3 (7.6)
15 (38.4)
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White blood cells >12x10³/µL
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Signs and Symptoms
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premenarchal patients (39 cases)
ACCEPTED MANUSCRIPT Table 2 Abdominal ultrasound findings associated with ovarian torsion in 31 premenarchal patients (38 cases) N (%)
Any Abnormal finding
28 (73.7)
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Ultrasound Findings
11 (28.9)
Edema of the ovary
9 (23.6)
Enhanced echogenicity
1 (2.6)
Free pelvic fluid
3 (7.9)
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Enlarged ovary
Abnormal Doppler Flow
1 (2.6)
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Absence of venous flow Absence of arterial and venous blood flow
14 (36.8)
Whirlpool sign
2 (5.2)
Additional Findings Simple cyst
11 (28.9)
Complex cyst
4 (10.5)
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Normal abdominal ultrasound
10 (26.3)
ACCEPTED MANUSCRIPT Table 3 Additional pathologic findings at surgery and surgical treatment in 32 premenarchal patients with ovarian torsion
Findings and Procedure
N (%)
Affected Side 18 (46.1)
Left
21 (53.8)
Additional Pathologic Findings
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Right
25 (64.1)
Enlarged ovary (>4cm)
13 (33.3)
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Black-bluish ovary Simple ovarian/para-ovarian cyst
7 (17.9)
Complex ovarian/para-ovarian cyst
3 (7.6)
Hemorrhagic cyst
1 (2.5)
Hydrosalpinx
1 (2.5)
Appendicitis
3 (7.6)
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Inguinal hernia
3 (7.6)
Surgical Procedure Detorsion
20 (51.2) 9 (23.0)
Detorsion and cystectomy
5 (12.8)
Detorsion and fixation
6 (15.3)
Ovarian resection
2 (5.1)
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Detorsion and drainage
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(39 cases)