Endocrine Abstracts

Introduction: Signs of hyperandrogenism are frequent complaints for young adults and adolescents and a common cause of presentation to endocrinology service. Even if polycystic ovary syndrome is the most frequent etiology, differential diagnosis is mandatory. (1,2)

Case report: We present the case of a 26-year-old female patient, obese, smoker, with a personal history of polycystic ovary syndrome and a family history of diabetes mellitus, who presented to our endocrinology clinic for the investigation of secondary amenorrhea and recently installed hirsutism. Clinically, the patient presents menstrual irregularity (last menstrual cycle 8-9 month ago), hirsutism (Ferriman Gallwey score 16) and acne; BMI 32.05 kg/m². Biological assessment revealed hyperandrogenism (testosterone 263,5 ng/dl, androstenedione 20,347 ng/ml and 17-OH-Progesterone 29,361 nmol/l) while normal gonadotropins levels, presence of insulin-resistance (HOMA-IR 3,7) and normal values of DHEAS, Prolactin, Estradiol, beta-HCG, inhibin. Cortisol hypersecretion/insufficiency was excluded and the thyroid function was normal. Transvaginal ultrasound identified the left ovary transformed into a tumor mass, inhomogeneous, hyperechoic, well-defined, well vascularised, measuring 4.4/4.2/4.8 cm. Laparoscopic excision of the tumor was performed. Histopathological and immunohistochemical examination confirmed a benign ovarian steroid cell tumor (NOS), with lipid rich cytoplasm (mitotic index 2/10< HPF, Ki67 4%, diffuse positivity for inhibin, calretinin and focal positivity for Melan A). Postoperatively, androgens values ameliorated - testosterone and androstenedione normalized (29,10ng/dl and 1,8ng/ml), 17-OH-Progesterone decreased but still mildly high (7,575nmol/l), the rest of the blood results being normal and clinically - menstrual cycles regulated without significant hirsutism amelioration and the patient lost weight (BMI 29,9 kg/m²).

Conclusion: Ovarian steroid cell tumours NOS, even if are rare, should be considered in any case of hyperandrogenism and excluded if elevated androgens levels are present. We identified a rare cause of hyperandrogenism in this patient even though many other causes were possible. Due to the fact that our patient wants to remain pregnant, it was decided to perform only the resection of the tumor, with the preservation of ovarian tissue.

References: 1. Jiang, W., Tao, X., Fang, F.et al. Benign and malignant ovarian steroid cell tumors, not otherwise specified:case studies, comparison, and review of the literature.J Ovarian Res 6,53(2013).2. Veilleux-Lemieux M, DiVasta AD. Severe hyperandrogenemia and insulin resistance in a 12-year-old girl. J Pediatr Adolesc Gynecol.2012 Aug;25(4):e99-101. doi: 10.1016/j.jpag.2012.04.002. PMID: 22840945.