An unusual example of mammary myofibroblastoma in a 36-year-old woman was reported. The central portion resembled mammary myofibroblastoma or solitary fibrous tumor of other sites and showed reactivity for CD34 and muscle-specific actin, with few cells staining for desmin. The peripheral portion resembled leiomyoma and showed strong reactivity for actin and desmin but only focal staining for CD34. We interpret this tumor as mammary myofibroblastoma showing frank peripheral leiomyomatous differentiation. Although closely related to solitary fibrous tumor, mammary myofibroblastoma appears slightly different because of its consistent slant toward myoid differentiation in the form of actin and variable desmin reactivity as opposed to the rare and sparse immunohistochemical expression of myoid features by solitary fibrous tumors of other sites. This case demonstrates this phenomenon carried to an extreme, a feature not previously documented in mammary myofibroblastoma. It is of interest to note the modulation of the antigenic profile with the differentiation process, that is, loss of CD34 and gain of desmin accompanying the myoid differentiation.