A promising treatment for spontaneous ovarian hyperstimulation syndrome due to familial partial lipodystrophy: GnRH analogs combined with cyst aspiration.

Abstract

Purpose: To present a patient with familial partial lipodystrophy (FPLD) and polycystic ovary syndrome (PCOS) who was admitted with spontaneous ovarian hyperstimulation syndrome (OHSS)-like extremely enlarged ovaries, which was successfully treated using gonadotropin-releasing hormone analogs and abdominal cyst aspiration in combination.

Method: This is a descriptive case report of a single patient with FPLD and PCOS.

Results: Clinical improvement was achieved 6 months after therapy besides progressive reduction in total testosterone and DHEAS. Furthermore, there was a significant improvement in hyperinsulinemia and hypertriglyceridemia. Additionally, reduction in the size of ovarian cysts, reduction in the size and number of localizations of acanthosis nigricans, reduction in scores of mFGS, and weight loss were also observed.

Conclusion: Although there are few reports in the literature describing the association between PCOS with FPLD, management of this novel spontaneous OHSS-like condition has not yet been clearly defined. In the case of extremely enlarged multicystic ovaries and severe hyperandrogenemia, GnRH analogs may be considered to prevent ovarian enlargement and reduce hyperandrogenemia, especially when other treatment options are inappropriate.