An unusual hormone pattern in a virilized woman affected by Sertoli-Leydig cell tumor. Report of a case.
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.
Satta
M A
MA
Institute of Endocrinology, University School of Medicine, Chieti, Rome.
De Rosa
G
G
Scoppola
A
A
Rabitti
C
C
Piccinno
E
E
Monaco
F
F
D000328:Adult; D005260:Female; D007987:Gonadotropin-Releasing Hormone; D006728:Hormones / Q000097:blood; D006801:Humans; D007984:Leydig Cell Tumor / Q000097:blood / Q000150:complications / Q000473:pathology*; D010051:Ovarian Neoplasms / Q000097:blood / Q000150:complications / Q000473:pathology*; D012707:Sertoli Cell Tumor / Q000097:blood / Q000150:complications / Q000473:pathology*; D013739:Testosterone / Q000097:blood; D014770:Virilism / Q000097:blood / Q000209:etiology*
D006728:Hormones; D007987:Gonadotropin-Releasing Hormone; D013739:Testosterone