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A case of ovarian Leydig cell tumor associated with adenocarci-noma of the endometrium in a 66-year-old woman is described herein, the eighth such case published. Clinically, both masculinizing and feminizing symptoms were observed: an increase in facial hair growth, slight baldness, clitoromegaly, and postmenopausal genital bleeding. Three biopsies of the endometrium during a 5-month preoperativc period showed atypical hyperplasia. Surgically resected material contained a Leydig cell tumor of the left ovary and focal adenocarcinoma in atypical hyperplasia of the endometrium. Serum levels of androgens and estrogens measured by radioimmunoassay decreased after removal of the tumor. Immunohistochemical studies revealed that the Ley-dig cell tumor contained testosterone, estrogens, and 17β-hydroxysteroid dehydrogenase (HSD) in the cytoplasm. This is the first report of a Leydig cell tumor in which the localization of 17β-HSD was demonstrated immunohisto-chemically.