In 2014, a 35-year-old male resident of Rio de Janeiro was admitted with ulcerated lesions on his nasal septum, nasal cavity, palate, and balanopreputial sulcus (Figure 1). The patient had a past history of alcohol and cocaine abuse. The lesions initially appeared in 2013 as erythematous and painful papules that grew progressively larger. Additional symptoms included headache, nasal obstruction, asthenia, and weight loss. The initial differential diagnoses included MCL, paracoccidioidomycosis, neoplasia, granulomatosis with polyangiitis, and tuberculosis. The diagnosis of MCL was confirmed on the basis of positive IgG and IgM serology and histopathological examinations of the nasal mucosa and larynx. Nasal mucosa histopathological analysis showed dermal infiltration of histocytes, lymphocytes, and plasma cells in addition to some dilated vessels. The histopathologic analysis of the larynx showed extensive ulceration and inflammatory infiltrates, and a single macrophage with amastigote-like form in the cytoplasm. Videolaryngoscopy showed granulomatous and infiltrative lesions in the hypopharynx and larynx, hyperemia, and swollen vocal folds. There were also infiltrative lesions on the epiglottis, pyriform sinuses, and false folds. Results of viral and fungal serological tests, direct mycological examination, borehole azimuthal acoustic reflection imaging (BAAR), chest tomography, and respiratory function tests were all negative. Treatment with meglumine antimoniate was started but discontinued on day 22 due to phlebitis and fever. The patient was discharged on regression of all lesions and was referred for outpatient follow-up, but he did not return. He returned in 2018 because of recurrence and worsening of his condition. He presented with right-side nasal collapse and an erythematous lesion, which extended from the filter to the interior of the nasal orifice (Figure 2). Additionally, he had sinusitis with a foul-smelling discharge and blood clots. The sinus tomography showed nasal septum perforation and mucosal thickening of the maxillary sinus, with no other alterations on rhinoscopy. A 21-day treatment with intravenous amoxicillin/clavulanate was initiated.
Serology for leishmaniasis was IgG-positive and IgM-negative and non-reactive for paracoccidioidomycosis. Nasal mucosa histopathology showed ulcerated squamous mucosa with seromucous glands in the chorion. There were marked acute and chronic inflammatory processes with a giant cell reaction (Langhans cells) and epithelioid histocytes, compatible with a diagnosis of leishmaniasis (Figure 3). Nasal fragment polymerase chain reaction (PCR) analysis using the primer set 120 base pair detected leishmaniasis. However, PCR examination did not allow for species evaluation. The patient was administered amphotericin B treatment for 14 days, after which his lesions improved, and he was discharged. He was re-evaluated three months later with continued healing and was followed-up by our outpatient service.