Superficial leiomyosarcomas are rare soft tissue tumors, most frequently involving the lower extremity. Leiomyosarcomas which could be misdiagnosis or delayed are very rare in the head and neck region. In this report, a 46-year-old man with postauricular leiomyosarcoma was presented. The tumor was totally excised with 3 cm margins. Immunohistochemically, the specimen showed reactivity for smooth muscle actin, vimentin, and desmin. No complications developed in the postoperative period and the patient was free of recurrence at the 26 months follow-up. Immunohistopathological examination is essential for an accurate histological diagnosis. The ENT surgeon should pay attention to superficial leiomyosarcoma with its specific behavior and treatment while evaluating the lesions in the head and neck region.