A 42-year-old woman with an epithelioid granular cell leiomyoma of the right breast is reported. The cells were spindle to polygonal and displayed finely granular cytoplasm. The smooth muscle differentiation of this tumor was shown by the immunohistochemical positivity of the neoplastic cells with anti-alpha smooth actin and antidesmin antisera. Microfilaments with focal densities were present in the cytoplasm at an ultrastructural level. The granular cytoplasmic changes are related to a relevant number of lysosomes within the neoplastic cells.