An Unusual Hormone Pattern in a Virilized Woman Affected by Sertoli‐Leydig Cell Tumor

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 dehydroepiandroste-ronesulfate (DHEA-S) (2,070 ± 6 ng/ml), androstenedione (1.8 ± 0.5 ng ml) and sex hormone-binding globulin (SHBG)(42 ± 3nM/L); there was normal urinary 17 keto-steroid (17-KS) excretion (11.7 mg 24 h), low urinary estrogen (E) excretion (3 + 0.4//g 24 h), suppressed basal gonadotropin concentrations (LH 0.9//III ml; FSH 3.2,uUI/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 hypothalamo pituitary axis against an endocrinological background of notable alteration of the androgen/estrogen ratio, where the androgens were slightly increased and the estrogens greatly reduced. Acta Pathol Jpn 39: 755-758, 1989.