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Cutaneous leishmaniasis (CL) due to Leishmania major (L. major) is common in the Middle East; however, this skin infection may be under-diagnosed when it presents atypically.To highlight the occurrence of uncommon presentations of CL that may elude diagnosis.A retrospective study was performed among patients who presented at The Sheba Medical Center between 2005 and 2014 with atypical clinical presentations of CL due to L. major.Twelve patients with unusual clinical presentations of L. major CL were identified. All infections were acquired in L. major – endemic areas of Israel. The average age was 37 years. The average number of lesions was 2. Nine patients presented with a form that mimicked other forms of CL, such as lupoid, giant ulcer, sporotrichoid and recidivans, and three had a variant resembling other infectious skin diseases, such as erysipeloid and verruciform. All patients required systemic therapy.Cutaneous leishmaniasis due to L. major can masquerade as many other infectious and inflammatory diseases. In addition, it can mimic clinical forms of New World CL. We suggest that in endemic countries or in travellers returning from countries where L. major is endemic, polymerase chain reaction (PCR) for Leishmania-specific DNA should be performed routinely in cases of unusual presentations of dermatitis with a single or a few lesions, even if a diagnosis of CL was not considered by the referring clinician.