A 24-year-old woman was admitted to hospital because of
hirsutism,
virilism and
amenorrhea, which had appeared 6 months earlier. Endocrinological evaluation showed a slightly elevated serum level of
testosterone (1.2 +/- 0.05 ng/ml), normal plasma levels of dehydroepiandrosteronesulfate (
DHEA-S) (2,070 +/- 6 ng/ml),
androstenedione (1.8 +/- 0.5 ng/ml) and
sex hormone-binding globulin (SHBG)(42 +/- 3 nM/L); there was normal urinary 17-ketosteroid (17-KS) excretion (11.7 mg/24 h), low urinary
estrogen (E) excretion (3 +/- 0.4 micrograms/24 h), suppressed basal
gonadotropin concentrations (LH 0.9 microUI/ml; FSH 3.2 microUI/ml) and an exaggerated response to the
LH-RH test. At
laparotomy, a monolateral ovarian
tumor was found, which was proved histologically to be a
Sertoli-Leydig cell tumor. After
tumor ablation, a regular menstrual cycle followed and progressive reduction of
virilism was noted. This was followed within 4 months by complete normalization of LH, FSH,
estrogen and
progesterone serum levels. The responsiveness to
LH-RH also became normalized. Two years after this operation, the patient had a normal pregnancy. This case of
virilization in a woman affected by a benign
Sertoli-Leydig cell tumor was primarily characterized by an unusual response of the hypothalamopituitary axis against an endocrinological background of notable alteration of the
androgen/
estrogen ratio, where the
androgens were slightly increased and the
estrogens greatly reduced.