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A case of endometrial carcinoma arising in a large adenomyomatous polyp is described in a 75-year-old woman. The malignant element appeared to be a mixed adenosquamous carcinoma that was embedded in diffuse adenomyomatous infiltration. The lesion was entirely intramural, and the surface endometrium was atrophic and not neoplastic. The absence of endometrial involvement led to a 6-year delay before hysterectomy was carried out. Pathologic study of the radically excised uterus showed evidence of metastasis to one paraaortic lymph node. Since this appears to be the first example of such an event, the English literature was reviewed for the associated findings of adenomyosis and carcinoma in an intramural setting with noninvolvement of the surface endometrium.