American cutaneous leishmaniasis, an endemic and increasing disease in Brazil, can present as skin ulcers and lesions in the mucous membranes of the nose, mouth and pharynx. Pentavalent antimony is the first choice drug for treatment, with a less favorable response of the mucosal forms. The difficulty of diagnosing and treating a case of mucosal leishmaniasis in a five-year-old child, with negative parasitological and immunological tests and polymerase chain reaction is emphasized. Only after several attempts was the smear test positive. The patient developed persistent secondary bacterial infection on the lesions, lack of response to specific drugs and antibiotics and evoluted to septicemia and death.
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