Leishmaniasis is prevalent in most warm-climate areas of the world. We describe a long-undiagnosed case of mucocutaneous leishmaniasis in Italy that might have been contracted in Costa Rica. The patient's signs and symptoms included granulomatous-like lesions on the forehead and legs, nasal obstruction accompanied by serous and crusted rhinorrhea, and diffuse granulomatous-like lesions in the septum and turbinates. The patient was treated with three on-off cycles of itraconazole. At the 1-year follow-up, all his lesions had nearly disappeared