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Cutaneous leishmaniasis (CL) caused by Leishmania major is common in southern Israel, while Leishmania infantum (sub-strain of L. donovani, causing zoonotic visceral leishmaniasis) infections were rarely reported in Israel and only in other regions. We report the first case of L. infantum infection in southern Israel, presented atypically as CL in an immunosuppressed 47-year old male. The patient was treated with liposomal amphotericin-B and recovered, without extra-cutaneous complications. Diagnosis of L. infantum CL was confirmed by microscopic identification of amastigotes in Gimsa-stained smear of skin lesion, positive blood serology and a positive polymerase chain reaction (PCR) amplification of the internal transcribed spacer 1 genes (ITS1) and restriction fragment length polymorphism (ITS1 PCR-RFLP). We also review the medical literature on old-world CL caused by L. infantum. Multiple L. donovani/infantum CL cases were identified in the literature search. These can be divided schematically to two: 1) In several endemic countries, L. infantum strains are the main causative agents of CL; 2) In other regions, CL is almost exclusively caused by L. major or L. tropica, while L. donovani strains CL cases were reported sporadically or as imported disease.