We describe a case of isolated laryngeal leishmaniasis in an 55-year-old smoker, treated with steroids for rheumatoid arthritis, in the absence of concomitant visceral or cutaneous localizations. Clinical presentation was dominated by dysphonia. Laryngeal biopsy revealed the presence of Leishmania amastigotes, which were characterized by species-specific polymerase chain reaction as L. donovani parasites. In endemic areas, Leishmania infection may present with atypical localizations and it has to be considered as a possible cause of laryngeal symptoms, especially in subjects with known immunosuppressive diseases or under treatment with immunosuppressive drugs.