Abstract

Gonadotroph pituitary adenoma (Gn-PA) may rarely cause ovarian hyperstimulation syndrome (OHSS) leading to infertility in women, though this remains poorly described. We present a quantitative systematic review including 2 institutional and 48 literature cases with Gn-PA causing OHSS to thoroughly describe the clinical features and therapeutic outcomes from multidisciplinary aspects. The patients demonstrated a mean age of 31.5 years and a mean FSH of 14.4 IU/L. Estradiol was high at > 350 pg/mL in 82%. The mean maximal adenoma diameter was 22 mm with Knosp grade being ≥ 3 in 10 cases. Abdominal surgery preceded adenoma resection in 24 (48%). Among 25 for whom extent of resection was recorded, total adenoma resection was achieved in 12. Through a mean follow-up of 25 months, adenoma recurrence was observed in 5, which were treated with re-resection (2), radiation (2), and medical therapy followed by bilateral oophorectomy (1). Medical therapies were partially effective or ineffective, and adenoma shrinkage did not follow; gonadotropin-releasing hormone (GnRH) agonists/antagonists were partially effective in 20% (2/10), dopamine agonists in 44% (8/18), and somatostatin analogues in 50% (1/2). Four experienced swelling of tumor/ovaries after GnRH agonists/antagonists administration. Overall, chemical remission was obtained in 26 of 28, normalization of ovaries in 25 of 27, and successful pregnancy in 12 of 14. Adenoma resection is the main treatment, leading to reduction in ovarian size and biochemical remission, with a high likelihood of subsequent spontaneous pregnancy. Increased awareness of this rare condition may help avoid unnecessary abdominal procedures.

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