Lupoid cutaneous leishmaniasis (LCL) is a rare, atypical presentation of cutaneous leishmaniasis (CL). In this report, the authors present the case of a severe LCL in an elderly patient who presented to our dermatology department with severe, painful ulcerated lesion on his midface with cosmetic deformity to his nose. He had a history of CL 3 years ago at the same place. Histopathologic examination showed epidermal and dermal changes with chronic inflammatory infiltrate and no leishmaniasis bodies were detected. He was admitted and treated with systemic glucantime (60 mg/kg) for a month followed by hydroxychloroquine (200 mg twice a day) for another month with favorable outcome. Countries with endemic CL should consider LCL in patients with a history of leishmaniasis and a similar clinical presentation, especially that it could be misdiagnosed with other granulomatous cutaneous conditions, thus leading to cosmetic deformities that can be avoided with early adequate treatment.

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