Adenomyoepithelioma of the breast is an uncommon tumor characterized by the presence of both epithelial and myoepithelial cells; its first full description was published in 1970 by Hamperl. A 45-year-old woman presented a left breast lump that had been palpable for 4 weeks. There was no family history of breast cancer. Neither axillary nor supraclavicular lymph nodes were palpable. Craniocaudal mammography showed a 1.6 cm, well-defined nodule with several punctate intranodular calcifications in the subareolar region of the left breast. Color Doppler sonogram showed an ovoid, well-defined, homogeneous hypoechoic subareolar nodule with prominent peripheral vessels in the 6 o'clock position of the left breast. Operation was arranged and intraoperative frozen section examination revealed proliferation of round, oval or tubular glandular elements with intervening islands and bands of polygonal myoepithelial cells. Adenomyoepithelioma of the breast was the impression. Wide excision with adequate removal of the tumor and preservation of the left breast contour was successfully achieved. The diagnosis of adenomyoepithelioma of the breast was confirmed by immunohistochemical studies.