Pansky, Moty MD; Smorgick, Noam MD, MSc; Herman, Arie MD; Schneider, David MD; Halperin, Reuvit MD, PhD
Most cases of adnexal torsion in postmenarchal women are associated with underlying adnexal pathology,1–6 whereas the twisted adnexa do not have any visible morphologic pathology and appear otherwise normal in about 8–18% of cases.1–5 The condition is more common in premenarchal females (children or premenarchal adolescents) in whom torsion involving previously normal adnexa may constitute up to 15–50% of adnexal torsion cases.6–8 Earlier adnexectomy methods for treating torsion of normal adnexa in premenarchal females have been replaced by a laparoscopic adnexa-sparing procedure, referred to as adnexal detorsion.8 The newer approach conserves the adnexa, but may enable repetitive torsion events to take place.9–13 Most reports of recurrent torsion, however, were single case reports or small case series, and there appear to be no studies that attempted to evaluate the actual rate of torsion recurrence.
The literature on the possibility of recurrence, prevalence, and prevention of torsion of normal adnexa in postmenarchal and adult women is even more limited and includes only a few case reports.14 The aim of the current study was to investigate whether postmenarchal women are at risk for recurrent torsion of normal adnexa by comparing the retorsion rates of normal adnexa with those of pathologic adnexa.
MATERIALS AND METHODS
This retrospective study included all postmenarchal women with surgically diagnosed adnexal torsion who underwent surgery at our institute between January 2002 and April 2006. The cases were identified by a search of a computerized database for coding of “torsion.” Sixteen pregnant patients were excluded from the analysis, and the medical records of the remaining 62 patients were retrieved to extract information on demographic characteristics, previous torsion events, preoperative pelvic sonograms, intraoperative findings, and pathologic diagnosis.
After reviewing the medical records, the patients were classified into two groups according to the adnexal pathology. The first group included patients with any underlying adnexal pathology (ie, functional ovarian cysts, ovarian tumors, paraovarian cysts, and other tubal abnormalities), and the second group included patients with torsion of apparently normal adnexa. This classification was based on the pathologic diagnosis (where available) and on the combination of clinical data, preoperative ultrasound scans, and intraoperative findings in all other cases. In addition, all 12 patients with twisted normal adnexa received a postoperative ultrasound examination, either at our ultrasound unit (n=5) or at outpatient facilities (n=7). We obtained the results of the latter scans from a telephone interview with the patients. All repeat scans were performed within 2 months to 2 years after the last surgery, and all were consistent with normal adnexa. We could, therefore, place patients in the “torsion of previously normal adnexa” group when no enlargement, suspicious adnexal masses, or cysts were evidenced on preoperative imaging modalities, intraoperative inspection, and postoperative sonograms.
The recurrence of torsion in the two groups (normal versus pathologic adnexa) was evaluated by a telephone questionnaire, completed for 57 of the 62 study patients (90.5% compliance). The patients were asked whether they had undergone any additional surgery for adnexal torsion. Overall, we identified 11 patients (from both groups) who had recurrent torsion events. Nine of them underwent the second surgery in our department, and the relevant details were retrieved from the patients' medical records. The other two women (one with recurrent torsion of normal adnexa and the other with recurrent torsion of pathologic adnexa) underwent surgery at other institutions. A written summary of the surgical information was available for the former, but only self-reported general knowledge was available for the latter.
The surgical procedures included 1) detorsion alone, 2) detorsion followed by aspiration of the adnexal cyst, 3) detorsion combined with resection of the adnexal pathology (either cystectomy for ovarian/paraovarian cysts or salpingectomy for tubal pathology, such as hydrosalpinges), and 4) resection of the whole adnexa. For descriptive purposes, the first two were considered collectively as “minimal operations.”
The postmenarchal patients were divided into three age groups: adolescents (16–19 years), reproductive-aged women (20 years to menopause), and postmenopausal women. The age cutoff (19 years old) between adolescence and womanhood was used in accordance with the definition of the World Health Organization.
Statistical analysis was performed with the WINPEPI software (Available at: http://sagebrushpress.com/PEPI.html). The Mann-Whitney rank sum test and the Fisher exact test were applied as appropriate. The torsion recurrence rates in the two groups were compared with the Kaplan-Meier survival analysis. P<.05 was considered statistically significant. The study was approved by the Assaf Harofe Medical Center Institutional Review Board.
Sixty-two postmenarchal women diagnosed with adnexal torsion were included in the study. The women were divided into two groups: those with normal adnexa and those with pathologic adnexa. Twelve women (19.4%, 95% confidence interval [CI] 0.1–0.3%) were placed in the torsion of normal adnexa group and 50 (80.6%, 95% CI 0.7–0.9) in the torsion of pathologic adnexa group. The former were younger (25.5±7.9 versus 31.4±11.4 years, P=.10) and had a lower median parity (0, range 0–3, versus 1, range 0–5, P=.06). There were no significant proportional age differences or differences in the numbers of women with previous abdominal or pelvic surgery (33.3%, 95% CI 0.1–0.6 versus 14%, 95% CI 0.06–0.3, respectively, P=.20) between the two groups (Table 1).
The clinical presentation did not differ between the normal and the pathologic adnexa groups: abdominal pain was the most prevalent symptom (100%, 95% CI 0.8–1, and 94.3%, 95% CI 0.8–1, respectively, P=1.0), followed by nausea and vomiting (73.3%, 95% CI 0.5–0.9, and 47.2%, 95% CI 0.3–0.6, respectively, P=.09) and by subfebrile range fever (20%, 95% CI 0.05–0.4, and 3.8%, 95% CI 0.006–0.1, respectively, P=.07). Most cases of torsion in the two groups occurred on the right side (66.7%, 95% CI 0.4–0.9, and 56.6%, 95% CI 0.4–0.7, respectively, P=.6). The prevalence of an elevated white blood cell count (defined arbitrarily as more than 11,000 cells/mL, which was present in 26.7%, 95% CI 0.09–0.5, and in 33.9%, 95% CI 0.2–0.5, P=.7), and the interval between admission to surgery (18.2±24.9 versus 17.5±26.2 hours, respectively, P=.90) were also similar for the two groups.
The torsion recurrence rates were determined for the 57 women with known outcomes after a median follow-up period of 2.6 years (range 0.2–24) in the twisted normal adnexa group and 2.6 years (range 0.3–7) in the twisted pathologic adnexa group. The recurrence rates were 63.3% (7 of 11, 95% CI 0.3–0.9) compared with 8.7% (4 of 46, 95% CI 0.02–0.2, P<.001), respectively, with a post hoc power analysis of 93.8%. The Kaplan-Meier survival analysis was also statistically significant (log rank=7, P=.008).
Among the seven women with recurrent torsion of normal adnexa, the second torsion event involved the ipsilateral side in four women (57.1%, 95% CI 0.2–0.9) and the contralateral side in three women (42.9%, 95% CI 0.1–0.8), and the median interval between the two events was 2 years (range 0.3–24 years). The median age of these seven patients was 17 years (range 0.5–24) at the first torsion event and 23 years (range 17–27) at the second torsion event, and all but one were postmenarchal at the time of the first torsion event. The second torsion event also involved normal adnexa and was managed in three of the cases by fixing the adnexa to the pelvic sidewall (ovariopexy). None of the seven patients had a third torsion event during the follow-up period.
The retorsion rate in the group of women with twisted pathologic adnexa differed according to the surgical management of the first torsion event (Table 2). The recurrence rates were higher in the minimally operated patients (including detorsion both with and without cyst aspiration) compared with patients who underwent a resection of the adnexal pathology or a resection of the whole adnexa (20%, 95% CI 0.04–0.5, versus 5.3%, 95% CI 0.001–0.3, and 0%, respectively), although this difference did not reach a level of statistical significance (P=.2).
Adnexal torsion had traditionally been managed by removal of the adnexa,1 because of the concern about thrombus formation in the ischemic ovarian vein, which could give rise to thromboembolic events. Later on, a conservative surgical approach was proposed, in view of the minimal risks of thromboembolism and of the potential for follicular activity regeneration.15 Adnexal detorsion has also replaced the earlier adnexectomy methods in premenarchal females,8 but because many cases of torsion in these youngsters (up to 50%) involve otherwise normal adnexa, it has enabled repetitive torsion events to take place.6–8 Several case reports and small case series have indeed described the clinical presentation of repeated torsion of normal adnexa in premenarchal females,9–13 but these observations are too limited in scope for the purposes of determining the rate of retorsion or its risk factors.
Torsion of normal adnexa is considered uncommon among postmenarchal women in whom its prevalence is reported as being from 8% to 18%1–5 of all torsion cases. Women with twisted normal adnexa constituted 19.4% of the torsion cases that presented to us, and the disparity in the prevalence may be explained by the different methods of patient selection used in the previous and current studies. Previous case series also included women with torsion during pregnancy (up to approximately 25% of their study population), where torsion is usually a result of functional ovarian cysts combined with radical anatomical changes in the pelvis due to the enlargement of the uterus.1,3–5 This combination would increase the relative prevalence adnexal torsion with underlying pathologies and reduce the relative prevalence of torsion of normal adnexa.
The existing literature on the subject of recurrent torsion of normal adnexa in postmenarchal women is sparse. Zissin14 described a 38-year-old woman with a recurrent torsion of the right normal adnexa.
Although the current study has limited power, and only 12 patients were included in the “torsion of normal adnexa” group, we still demonstrated a significant risk for retorsion, ie, approximately 60% compared with the approximately 8% retorsion risk for the pathologic adnexa. The danger of a repeat torsion event of the normal adnexa did not subside over time: one of the patients experienced retorsion 24 years after the first event. Furthermore, as with premenarchal females, both adnexa were at risk for retorsion, ie, the one ipsilateral and the one contralateral to the previously affected one.
The management of recurrent torsion of normal adnexa in premenarchal females is still a matter of controversy, especially the questions of whether the surgical management should include ovarian fixing procedures at all, whether these procedures should be performed at the time of the first torsion event, and whether one or both ovaries should be fixed in place. On the one hand, the serious sequelae of recurrent torsion and the potential decrease in reproductive capacity seem to justify ovariopexy—even bilaterally—at the first torsion event. On the other hand, the fixing procedures themselves may potentially endanger the reproductive system by (at least theoretically) interfering with adnexal blood supply, tubal function, or tuboovarian communication. Furthermore, because a prospective long-term follow-up of the patients is not yet available, there is no evidence that ovarian fixing reduces the retorsion rate compared with controls, and there are no definite data on its long-term complications. Until such data are available, most authors believe that prudent management of twisted normal adnexa in premenarchal females warrants bilateral ovariopexy, which may be performed at the initial emergent surgery or later on as an elective procedure.9–13,17
The management of postmenarchal women with twisted normal adnexa has not been discussed in the literature. The current study raises the same dilemmas as those faced for premenarchal females. Reproductive-aged women without a suspected malignancy should be offered an adnexa-sparing surgical procedure. Then, in view of the high retorsion rate and the possibility of contralateral recurrence, the addition of bilateral preventive ovariopexy (either at the same surgery or as a later elective procedure) should be discussed. We could not evaluate whether ovariopexy is preferable to detorsion alone in the prevention of recurrence because of the relatively few events in this report. As such, we do not have enough evidence upon which to base a definite recommendation. Our data, however, do provide a justification for informing patients about the risk of retorsion and the need for a close follow-up and prompt assessment should symptoms recur.
In conclusion, the conservative management of twisted normal adnexa in postmenarchal women may possibly predispose them to recurrent events involving both the ipsilateral and contralateral adnexa. Ovariopexy procedures may prevent further torsion events and should be considered in the management of these patients, but clinical proof of reduced risk of retorsion after ovariopexy awaits confirmation by additional investigations.
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