We present the first case of mucocutaneous leishmaniasis in Algeria, diagnosed in an immunocompetent 42-year-old man exhibiting an infiltrated and ulcerated plaque leading to macrocheilitis of the entire lower lip. He was a police officer who lived in a village in Ain El Hammam (Kabylie region, known as an active focus of zoonotic visceral leishmaniasis) without any history of travel for the previous 3 years. He suffered from cutaneous lesions for 22 months due to the misdiagnosis of a skin lesion resembling other diseases such as Crohn disease or sarcoidosis.
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