Leishmaniasis, as a vector-borne disease, is transmitted by sandfly and caused by Leishmania protozoa. Brain involvement rarely occurs in visceral leishmaniasis. In this paper, a rare case of pons involvement by visceral leishmaniasis (VL) is reported. A 54 yr old man from Southwest of Iran (Yasuj) presented to the Emergency Ward with a 3-wk history of headache (continuous, throbbing, and general), fever, chills, weakness, anorexia, and weight loss.