Virilization of a post-menopausal woman by a testosterone-secreting Leydig cell type adrenal adenoma

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Abstract
In a 60 yr old virilized woman the plasma testosterone concentration was markedly elevated, whereas the plasma cortisol and ACTH and urinary 17-ketosteroids, 17-hydroxycorticoids, pregnanetriol and dehydroepiandrosterone were normal. The plasma levels of lutropin and FSH were in the postmenopausal range. Dexamethasone suppressed the urinary 17-ketosteroids, 17-hydroxycorticoids and pregnanetriol normally, but had no effect upon the plasma testosterone. These findings led to the tentative diagnosis of an ovarian hilus cell tumor. Bilateral oophorectomy revealed bilateral hyperthecosis without a tumor and did not result in any decrease in the plasma testosterone level. An attempt of adrenal vein catheterization succeeded only on the left side. The lack of a gradient in the testosterone concentration between blood from the left adrenal vein and a peripheral vein prompted a surgical exploration of the right adrenal gland, which led to the discovery and removal of an encapsulated Leydig cell type adenoma, characterized by Reinke''s crystalloids. A testosterone concentration gradient between right adrenal and peripheral venous blood obtained intra-operatively, rapid normalization of the plasma testosterone concentration postoperatively, results of a tumor incubation study and the clinical outcome proved that this adenoma had been the source of the excessive androgen. A history of a late menarche and persistent menstrual irregularities together with elevated gonadotropins despite excessive testosterone pointed to the possibility of a long-standing gonadotropin-dependent gonadal cell population in the adrenal, which was fully activated only by the menopausal gonadotropin rise.