Case: We report a 32-year old relapse case of Visceral leishmaniasis, treated with Paromomycin who belonged from a endemic zone of Bihar state, India. After confirmation, he was treated with Amphotericin B, followed by Liposomal Amphotericin B in full course and even in higher dose. But after each therapy, the patient either did not responded or relapsed after treatment. Ultimately, the patient was successfully treated with combination therapy of Liposomal amphotericin B and Miltefosine without any relapse.
Conclusion: The multi-drug unresponsive Visceral leishmaniasis cases could pose a major threat to treatment strategy in the elimination program. In such situation, combination therapy seems to be a better approach that needs to be explored.