We report the first case of visceral leishmaniasis (VL) from the non-endemic western hilly region of Nepal. The patient presented with a history of high-grade fever, abdominal distension, anemia, and weight loss. The case was confirmed as VL by microscopical detection of the Leishmania species amastigote in bone marrow aspiration and by a positive result for the rK39 test. The patient was treated with 0.5-1.0 mg/kg of Amphotericin B for 14 days (total of 405 mg), and amastigotes were negative on discharge. Five months later, this patient again developed fever, abdominal distension, and anemia. Clinical and hematological examinations suggested a relapse of VL. The patient was treated with 1 mg/kg of Amphotericin B for 18 days (total of 515 mg) and was clinically improved on discharge.