BACKGROUND: Leishmaniasis is a zoonotic disease caused by unicellular protozoa of the Leishmania genus. The infection can spread through zoonotic or anthroponotic transmission, depending on the species, with the phlebotomine sandfly serving as the primary vector. Leishmaniasis is endemic in tropical regions of Asia and Africa. While mucocutaneous leishmaniasis is rarely reported in Ethiopia, both the cutaneous and visceral forms of the disease are more commonly seen. The clinical spectrum of Leishmania infection includes visceral leishmaniasis (the most common form), as well as cutaneous, mucocutaneous, mucosal, and post-kala-azar dermal leishmaniasis. The mucosal form typically involves the nasal and oral mucosa, though in rare cases, it can also affect the laryngeal and pharyngeal mucosa. CASE PRESENTATION: This report discusses a case of laryngeal leishmaniasis presenting as hoarseness of voice and discomfort during swallowing, with a focus on clinical presentation, diagnostic process, and management. A 31-year-old Ethiopian man from Addis Ababa, Ethiopia, presented with a 6-month history of hoarseness and difficulty swallowing. He had a history of travel to Humera, a region endemic for leishmaniasis. Flexible nasolaryngoscopy revealed whitish erythema and irregular margins on the right vocal cord. Laryngeal cancer was initially suspected, and a microlaryngoscopy with biopsy was performed, which confirmed the presence of Leishmania amastigotes. He was treated with liposomal amphotericin B. After completing the treatment, his voice returned to normal, and repeat nasolaryngoscopy showed no abnormalities. CONCLUSION: Laryngeal leishmaniasis is a rare but important differential diagnosis for patients presenting with hoarseness, particularly those with a history of travel to endemic areas. Clinicians should consider leishmaniasis in the differential diagnosis of upper respiratory symptoms in endemic regions, even in the absence of classic skin lesions. Early diagnosis and appropriate treatment with antifungal agents such as liposomal amphotericin B can lead to full recovery, as demonstrated in this case.