Leishmaniasis is a disease caused by the intracellular protozoan parasite Leishmania and are known more than 20 species(1) harmful for men. A 74-year-old man, with sarcoidosis treated with methotrexate and corticoid, was assessed, in 2021, by an ENT specialist due to dysphagia, dysphonia, and odynophagia with a 5-year evolution and progressive worsening. A biopsy of the right vocal cord and epiglottis was performed, and the histology demonstrated the presence of amastigotes in the tissues coloured by Giemsa making the diagnosis of Leishmaniasis. The patient was referred to the Infectious Diseases Department, with the diagnosis of mucosal leishmaniasis, and hospitalized for treatment with Liposomal Amphotericin B. The dysphagia and odynophagia improved and was discharged to Infectious Diseases Day hospital to continue treatment. He completed a total of 10 days of treatment and continued follow up in Infectious Diseases, Pneumology and ENT departments. During this time the patient stopped treatment with methotrexate but maintained deflazacort 6 mg per day. In 2023, the patient presented with worsening dysphonia and dysphagia. A new biopsy of the epiglottis was performed in the ENT department. Leishmania DNA was detected, and histology was compatible with Leishmaniasis of the left larynx. He was hospitalized in Infectious Diseases department and started treatment with Liposomal Amphotericin B. The patient completed a total of 10 days of treatment, and, by this time, the medical team decided to maintain suppressive therapy once a month with Liposomal Amphotericin B, until the patient present with a CD4 leucocyte count superior to 350/mm³ . By the time of this article, the patient maintained follow up in the Infectious Disease department with monthly sessions of therapy.