A paradoxical inhibition of androgenic hyperproduction by a Sertoli-Leydig cell tumour ovary.
A 17 year old woman was evaluated for amenorrhoea and severe hirsutism (Ferriman-Gallway index = 31). Pelvic ultrasound demonstrated a right unilateral ovarian mass (6 x 5 cm), whereas the computed tomography for the adrenal gland was normal. Endocrinological findings revealed normal concentrations of oestradiol, progesterone, dihydroepiandrosterone sulphate, cortisol, prolactin, follicle-stimulating hormone, luteinizing hormone and adrenocorticotrophic hormone (ACTH). Total testosterone, free testosterone, androstenedione and 17-hydroxy-progesterone concentrations, already elevated at basal conditions, did not increase after an ACTH test, whereas they decreased significantly after dexamethasone administration and increased after a human chorionic gonadotrophin test. Of all the tumour markers investigated, tissue polypeptide antigen and alpha 1-fetoprotein showed an increase in concentration. Selective venous ovarian catheterization indicated the presence of an androgen-producing tumour in the right ovary. The histopathological diagnosis was consistent with a Sertoli-Leydig cell tumour ranking between an intermediate and a poor grade of differentiation, with heterologous elements characterized by mucinous epithelium of the gastro-intestinal type. An endocrine evaluation performed postoperatively showed a normalization of all serum pathological hormones and tumour markers studied. Some particular aspects were focused on and discussed.
Amato
G
G
Department of Endocrinology, Faculty of Medicine and Surgery, Second University of Naples, Italy.
Izzo
G
G
Izzo
A
A
D000293:Adolescent; D000568:Amenorrhea / Q000209:etiology; D000728:Androgens / Q000096:biosynthesis*; D014408:Biomarkers, Tumor / Q000097:blood; D005260:Female; D006628:Hirsutism / Q000209:etiology; D006728:Hormones / Q000097:blood; D006801:Humans; D007984:Leydig Cell Tumor / Q000150:complications / Q000378:metabolism* / Q000473:pathology; D010051:Ovarian Neoplasms / Q000150:complications / Q000378:metabolism* / Q000473:pathology; D012707:Sertoli Cell Tumor / Q000150:complications / Q000378:metabolism* / Q000473:pathology
D000728:Androgens; D014408:Biomarkers, Tumor; D006728:Hormones