Abstract Angioleiomyomas (ALMAs) are benign tumors that arise from smooth muscle cells that form the tunica media of vascular structures. To our knowledge, only five cases of ALMA of the external auditory canal (EAC) have been reported. We report the case of a 52-year-old man with left otalgia, otorrhea, auricular fullness, and hearing loss. On otomicroscopic examination, a reddish and smooth mass completely occluding the EAC was seen. Computed tomography scan showed a soft tissue mass with a complete opacification of the left EAC. A possible diagnosis of ALMA should be considered when finding a tumor inside the external ear canal. An accurate pathological examination should be performed to differentiate benign vascular ALMA from malignant leiomyosarcomas. Complete surgical excision is the treatment of choice in benign tumors. Cite this article as: Tombolini M, Ralli M, Greco A, de Vincentiis M, Carletti R. Leiomyoma of the External Auditory Canal. J Int Adv Otol 2019; 15(1): 169-72.