- Cutaneous leishmaniasis, caused mainly by Leishmania major, is endemic in southern Israel. It is characterized by multiple skin lesions on the skin's patient. The treatment often includes only topical treatment, and treatment failures are not uncommon. Liposomal amphotericin B, a drug approved for visceral leishmaniasis treatment, has rarely been used for the cutaneous disease, especially for Old World cutaneous leishmaniasis. We report a 1-year-old patient with multiple skin Lesions, diagnosed as leishmania major infection. The patient's parents refused topical treatment, as they were concerned regarding the possibility of treatment failure and residual facial scars. The patient was treated with intravenous liposomal amphotericin B, given as a 6 dose regimen and was cured clinically without any complications. Post-treatment evaluation, including direct microscopy, culture and polymerase chain reaction (PCR) revealed no evidence of residual disease. We believe that liposomal amphotericin B, although expensive, should be considered for cutaneous leishmaniasis treatment, when systemic treatment is needed, such as in cases with multiple facial skin lesions.