Cutaneous Leishmaniasis (CL) is a skin infection caused by various species of Leishmania parasites, which is transmitted by infected Phlebotomus sandfly bites. Pentavalent antimonials (meglumine antimoniate and sodium stibogluconate) are used for the treatment of adult CL patients as an effective and safe method. Liposomal amphotericin B is an alternative for the treatment of choice in cutaneous leishmaniasis cases which pentavalan antimony contraindicated or unresponsive to pentavalent antimony therapy. In this study, successful treatment with systemic liposomal amphotericin B of a cutaneous leishmaniasis case developing local side effects related both systemic and intralesional meglumine antimonate treatment was presented.