A 48 year old woman had a hysterectomy with bilateral salpingo-oophorectomy because of uterus myomatosus. Histologic examination of the surgical specimen revealed in one of the ovaries an adenocarcinoma in situ that had unequivocally developed within a focus of endometriosis. In addition, atypical epithelium was found in a small circumscribed area in one of the many islets of endometriosis in the myometrium.