Visceral leishmaniasis, a protozoan disease caused by Leishmania infantum, is endemic in the Mediterranean basin, especially southern and Tyrrhenian Italy. Its aetiological agent can also sporadically cause isolated laryngeal localization in at-risk patients (i.e., heavy smokers, immunocompromised patients). This rare localization is often pauci-symptomatic and thus can easily escape diagnosis. A case of isolated leishmaniasis limited to the left vocal cord in an immunocompetent Italian male without significant risk factors, randomly discovered upon histological examination, is described herein. We inquire how many patients affected by non-specific symptoms such as dysphonia and live in countries where Leishmania infantum infection is reported, could be truly affected by Leishmania spp infection.