(78) Management of Asynchronous, Recurrent, Bilateral Pre-Menarchal Ovarian Torsion

Background: Ovarian torsion (OT) is a surgical emergency with an incidence of 5-20 in 100,000 in the pediatric population. Among pre-menarchal patients, more than half of OT occurs in the absence of a cyst, which contributes to an unclear etiology or long-term management strategy. Here, a patient with recurrent, asynchronous, bilateral OT refractory to prior bilateral oophoropexy underwent novel management with a combined left three-point oophoropexy and right oophorectomy for ovarian tissue cryopreservation (OTC).

Case: An 11-year-old pubertal, pre-menarchal female had a first episode of right OT at 9 years 4 months of age, which was managed with laparoscopic right adnexal detorsion. Three months later, she had left OT and underwent detorsion. At age 10 years, she underwent interval laparoscopic bilateral oophoropexy at outside institution via shortening of the bilateral utero-ovarian ligaments. However, at age 10 years 5 months, she experienced repeat right OT despite prior oophoropexy and underwent laparoscopic right ovarian detorsion. In the setting of recurrent bilateral OT, she and her family elected to proceed with OTC of the right ovary and repeat oophoropexy of the left ovary via three-point technique. Following completion of a pelvic ultrasound which confirmed normal appearance and size of the bilateral ovaries, and demonstration of a normal anti-Müllerian hormone (AMH), she underwent laparoscopic right oophorectomy for OTC and left oophoropexy at age 10 years 7 months. Intra-operative findings showed failure of both prior oophoropexies. 6 months later, she has been in good health without signs of recurrent left OT.

Comments: Hypermobility of the ovarian ligaments may contribute to pre-menarchal OT. Management strategies have recommended adnexal detorsion with consideration of oophoropexy for recurrent OT. However, oophoropexy does not guarantee prevention of subsequent OT. As supported by this case, the right ovary is at increased risk of torsion. After counseling, the patient and her family elected to proceed with definitive management of the right ovary with right OTC and a previously reported 3-point oophoropexy technique on the left, avoiding the reported failure of shortening of the utero-ovarian ligament. Normal ovarian anatomy on pelvic ultrasound and confirmation of acceptable ovarian reserve with normal AMH level confirmed optimal pre-operative benefit. In conclusion, for optimal medical management and improvement in quality of life in select patients, recurrent, asynchronous bilateral ovarian torsion may be managed with unilateral OTC of the highest risk ovary and contralateral oophoropexy.