A 37-year-old man presented with dyspnea and erythematous, nodular facial lesions. An examination revealed nasopharyngeal edema, and imaging showed prevertebral swelling. An extensive infectious and autoimmune evaluation was negative. A cheek biopsy was done, and polymerase chain reaction was positive for Leishmania braziliensis, confirming the diagnosis of mucocutaneous leishmaniasis. This is a progressive manifestation of a parasitic infection by Leishmania spp. It may present several years following resolution of an initial cutaneous infection. In this case, 26 years. Negative serology for leishmaniasis does not exclude active disease, and a high index of suspicion leads to early diagnosis and treatment.