Background Lupoid cutaneous leishmaniasis (CL) is a rare form of CL having a striking resemblance to other granulomatous cutaneous conditions of infectious or inflammatory origin. The authors present a patient with a facial lupoid CL and discuss the diagnostic tools of this parasitological infection, the main differential diagnosis, and treatment. Case report A 54-year-old Tunisian woman, with no past medical history of lupus erythematosus or infectious disease, presented with a 3-month history of a slowly enlarging erythematous and infiltrated plaque, extending over the nose, the right cheek, and the internal aspect of the right lower eyelid. Microscopic examination of a parasitological smear showed numerous leishmania in their amastigote form, inside monocytes, confirming the diagnosis of CL. Clinical aspect was in favor of lupoid CL. The patient was cured by fluconazole 200 mg/day for 6 weeks after pancreatic intolerance with intramuscular meglumine antimoniate (60 mg/kg/day for 7 days), and no response to doxycycline (200 mg/day for 6 weeks). Discussion Lupoid CL is easily suspected in countries in which there is an endemic of leishmaniasis. In countries where there isn’t an endemic, although rarely observed, this diagnosis should also be kept in mind in front of an infiltrated facial lesion of a tuberculoid aspect on histology, especially when there is a positive travel history to an area in which there is an endemic.