Endocrine Abstracts

Introduction: Hyperthecosis ovarian is a type of hyperandrogenism and severe insulin resistance, usually found in postmenopausal women.

Case Report: This case is about a 12 year-old girl who came to our hospital because of a rapidly progressive virilization. She had no medical history. For the previous 10 months, she had felt progressive growth of terminal hair in androgen-dependent areas, abundant hair loss, facial acne and voice changes. Axilarche and pubarche had occurred when she was 10 years old. She had not had menarche. She was not in contact with exogenous testosterone preparations. Physical examination revealed a score of 28 on the Ferriman Gallway scale, mild cervical acanthosis nigricans, clitoris hypertrophy and Tanner stage 2 breast development. Laboratory tests showed: glucose 78 mg/dl, HbA1c 4.8%, FSH 7.47 (follicular phase 3.6–12.5 mU/ml), LH 10.33 (follicular phase 1.7–12.5), 21.47 prolactin (6–30 ng/ml), 17 OH-PG 2.8 (0.1–1.4 ng/ml), DHEAS 96.3 (33.9–280 mcg/dl) androstenedione 3.1 (0.5–4.7 ng/ml), total testosterone 4.23 (0.06–0.82 ng/ml), free testosterone 6.2 (0.2–3.2 pg/ml), SHBG 7 (20–140 nmol/l), basal cortisol 28 (6.2–19.4 mcg/dl), insulin 100.9 mIU/ml (2.6–24.9). Hypercortisolism and 21-hydroxylase deficiency tests were negative. The pelvic MRI showed enlarged ovaries with oval morphology of 50×17×18 mm (right) and 48×19×18 mm (left). The patient underwent a laparoscopic bilateral ovarian wedge resection. The pathology showed ovarian hyperthecosis with androgen-positive receptors. After the surgery, the total testosterone levels were 4.12 ng/ml. Treatment was initiated with flutamide 250 mg/day and metformin 2000 mg/day. The patient experimented the menarche two months after starting treatment. Currently it is being considered to start GnRH analogues and hormone replacement therapy with metformin.

Conclusions: The clinical interest of this case lies in the severity of the disease, early age of onset and the difficult therapeutic management of the disease in premenopausal women, with very little bibliography available.