In this case report, we describe a rare manifestation of amphotericin B (AMB) toxicity. A case of fever, hepato-splenomegaly and pancytopenia was diagnosed, based on serological test and demographic profile, as visceral leishmaniasis complicated with secondary haemophagocytic lymphohistiocytosis. He was managed with conventional AMB. Subsequently, patient was showing subjective and objective improvement. Suddenly after receiving 450 mg of cumulative dose of AMB, patient developed hearing loss. On evaluation, he was found to have bilateral mixed hearing loss. Patient was investigated for the causes of hearing loss. When nothing could be attributed as an aetiology, AMB was stopped (after 500 mg cumulative dose). After missing the dose of AMB, patient had a dramatic improvement in his sense of hearing, which was confirmed objectively by audiometry before and after the event.