Two cases of intralobular carcinoma of the breast showing myoepithelial cell differentiation are reported. One was an in situ lesion localized within a fibroadenoma; the second was predominantly in situ, but areas of invasion were present. The neoplastic cells had round to ovoid nuclei and were polygonal to spindle in shape displaying glycogen rich clear cytoplasm. Alpha-smooth muscle actin was present in the cytoplasm of the neoplastic cells in both cases. In one case the same cells displayed cytoplasmic microfilaments at electron microscopic level. Intralobular growth of neoplastic myoepithelial cells has never been described in the literature, and this line of differentiation has to be added to the endocrine and apocrine features occasionally observed in in situ lobular carcinomas of the breast.