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Original Research |

Syndrome of Benign Nodular Adrenal Hyperplasia Associated with Feminization and Hyperprolactinemia

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Grant support: NIH General Clinical Centers Grant (RR-53) and U.S. Public Health Service Grants CA 07304 and HL 14734.

▸Address reprint requests to Dr. Robert Boyar, Montefiore Hospital and Medical Center, 111 East 210 Street, Bronx, NY 10467.

Bronx, New York

Ann Intern Med. 1974;80(3):389-394. doi:10.7326/0003-4819-80-3-389
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A 38-year-old man with severe gynecomastia, hypogonadism, and impotence had a pituitary tumor associated with markedly elevated plasma prolactin concentrations. Testicular biopsy showed a spermatogenic maturation arrest, with decreased Leydig cells. Endocrine evaluation showed normal plasma and urinary gonadotrophins. Plasma estradiol and urinary estrogen excretion and production rates were normal but the plasma testosterone was 0.06 µg/100 ml, in the prepubertal range. The associated finding of bilateral nodular adrenal hyperplasia in this patient suggests that the adrenal cortex may be a target organ for the trophic action of human prolactin and, as well, may play an etiologic role in the development of adrenal tumors or hyperplasia or both. The clinical findings of "idiopathic" gynecomastia and infertility caused by spermatogenic maturation arrest could represent less severe forms of this syndrome.





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