Several ethiologics are responsible for submental region masses, among these dermoid cyst is the most common. In a patient with submental mass of 1.5× 2cm, the only clinical finding was a palpable mass. The result of excisional biopsy was reported as Leishmaniasis despite the fact that leishmaniasis can rarely manifest itself as a submental mass. After making diagnosis, the patient was referred to infectious disease service for further treatment.