A case of intrascrotal atypical leiomyoma is reported. This lesion is extremely rare and usually misdiagnosed. Microscopically, contain bizarre and pleomorphic tumor cells, as well as showed immunohistochemical evidence of smooth muscle differentiation. An important microscopic criterion in the distinction with other entity, as the leiomyosarcoma, is the number of mitotic figures, scarce in the atypical leiomyoma and high in the smooth muscle tumours malignant.