Cellular angiofibroma is a benign tumor of the superficial soft tissues of the vulvovaginal and inguinoscrotal regions of adult patients. Histologically, it is characterized by bland spindle cells arranged without any pattern in a stroma with thin collagen fibrils and evenly distributed small- to medium-sized blood vessels with hyalinized walls. The case of a 44-year-old female with a perineal cellular angiofibroma containing small leiomyomatous nodules is described in this case report. These nodules could probably be originated from smooth muscle differentiation of the spindle cell component of an otherwise conventional cellular angiofibroma. To our knowledge, the finding of distinct leiomyomatous nodules within a cellular angiofibroma has not been previously reported.