(17) Bilateral Antenatal Ovarian Torsion with Autoamputation: A Case Report

Background: While antenatal and neonatal ovarian cysts (OC) typically resolve spontaneously, a minority will result in torsion; when this occurs in utero, there is a higher risk of autoamputation (AA) than in postnatal patients due to delay in intervention. In one case series of 28 patients diagnosed with antenatal ovarian torsion (AOT), 60% had AA of the affected adnexa; however, all of them were unilateral. We present a case of an infant with bilateral AOT resulting in AA.

Case: IRB exempt status was confirmed through our IRB process. Antenatal ultrasound (US) at 33w1d found bilateral OC, with right simple cyst measuring 2 cm and left complex cyst measuring 4 cm. She was born at term via vaginal delivery. US on day of birth showed both OC stable in size and appearance; no normal ovarian tissue visualized. Tumor markers were normal range. She had normal external female genitalia. Repeat US at 1 week of life showed the left complex cyst stable, and the right cyst smaller. Repeat US at 1.5 months demonstrated the same complex cyst stable in size but now located on the right, and the previously seen right cyst resolved; no identifiable ovarian tissue. She was asymptomatic throughout. Due to persistence and complexity, decision was made to proceed with diagnostic laparoscopy, performed at age 3 months. Intraoperatively she was found to have a normal uterus but no identifiable left ovary or fallopian tube (FT), with a 4 cm cystic structure floating on the right but connected to the left infundibulopelvic (IP) ligament by a thin band of tissue, consistent with AA of the left adnexa. This was removed and on pathology there was no viable tissue. The right FT was truncated at the ampulla with no identifiable fimbria, no identifiable right ovary. Superior to the end of the right FT and above the pelvic brim, a 5 mm piece of white tissue was attached to the peritoneum on the side wall, not connected to the right IP or utero-ovarian ligaments, presumed to be what remains of the right adnexa. Given possibility of viable tissue, decision was made to leave this in place; biopsy was not attempted due to concern for damaging the small amount of tissue remaining.

Comments: Antenatal OC are becoming a more common finding with advances in antenatal US imaging, however the incidence of AOT is rare. Most literature reports on postnatal monitoring techniques and leans toward conservative management in most cases. Our case presents a unique situation in which earlier intervention might have outweighed the risk of surgery to potentially preserve ovarian tissue, and would suggest consideration for earlier surgical intervention in cases where bilateral adnexal masses are present.