Cutaneous leishmaniasis (CL) frequently occurs in many rural and urban areas of Iran. Leishmania major and L. tropica are principally two causative agents of CL in Iran. We report here a case of leishmaniasis of the ear in a 61-year-old man referred to the Reference laboratory, Kashan, central Iran, in Jan 2022. He suffered from a 2-month history of a 1×3 cm lesion on the left ear. In the microscopy examination, amastigotes forms of Leishmania spp. were observed. L. tropica was confirmed using a single PCR with species-specific primers. The patient was introduced to a physician to begin the treatment protocol. It is recommended that physicians, especially in an endemic area, investigate any atypical lesion for CL.