Endocrine Abstracts

Introduction: Severe hirsutism and virilisation, especially if occurring later in life and with rapid onset, should prompt the search for rare but potentially threatening causes such as an androgen secreting ovarian or adrenal tumour.

We present the case of a 47-year-old woman referred to our clinic with obesity, severe hirsutism, alopecia, acne, and deepening of the voice that appeared insidiously 2 years ago, along with amenorrhoea which she interpreted as menopause. Laboratory testing revealed polycythemia, impaired glucose tolerance, normal thyroid, IGF1, 17OH- progesterone, baseline ACTH, cortisol, and DHEAS levels with adequate suppression of cortisol after low dose (2×2 mg). Dexamethasone suppression testing (LDDST) excluding an adrenal hypersecretion. She had severe hyperandrogenism – total testosterone=6.38 ng/ml which increased paradoxically after LDDST (7.57 ng/ml) and normal gonadotropins and oestrogens (LH=2.63 mUI/ml, FSH=5.59 mUI/ml, and oestrogen=86 pg/ml) reflecting either androgen induced amenorrhea in premenopause, or testosterone mediated gonadotropin suppression after menopause. CT scans of the abdomen and pelvis and the ultrasound evaluation of the uterus and ovaries showed normal morphology – raising suspicion of ovarian hyperthecosis. The patient initially postponed surgery, so a short trial of GnRH agonist was attempted (triptorelin 0.1 mg s.c./month) with a small decrease in testosterone to 5.65 ng/ml after 1 month, further proving its ovarian origin. A repeat ultrasound showed a 1.94/1.65 cm mass in the right ovary, suggesting an androgen-secreting ovarian tumour. The patient underwent bilateral oophorectomy and total hysterectomy and the pathology report confirmed a benign Leydig cell tumour of the ovary. Testosterone levels normalized immediately post-surgery (0.30 ng/ml) and after 6 months the hirsutism and alopecia were significantly improved, haemoglobin levels normalized, but the obesity persisted and diabetes mellitus was diagnosed.

Conclusion: Although, typically large, Sertoli–stromal ovarian tumours can occasionally be small enough to avoid detection even by high-resolution imaging; in the presence of virilisation the differential diagnosis includes ovarian hyperthecosis. In either situation bilateral oophorectomy is recommended after the end of childbearing years.