We report the first case of visceral leishmaniasis (VL) relapse in a healthy individual after complete miltefosine treatment. The patient attended hospital with a history of fever for 2 months, splenomegaly, hepatomegaly, and weight loss. The case was confirmed as VL by microscopical detection of Leishmania parasites in a bone marrow specimen and by a positive result for the immunochromatography-based test targeting the Leishmania donovani rK39 antibody. A polymerase chain reaction (PCR) specific for the Leishmania kinetoplast minicircle gene was positive, and subsequent sequencing of the PCR-amplified product confirmed that this case was a L. donovani infection. The patient was treated with miltefosine for 28 days, during which time the response was good, and the Leishman-Donovan body (LD body) was negative on discharge. Ten months later, however, this patient again developed high fever and splenomegaly, and LD bodies and rK39 antibody were positive, thus indicating a relapse of VL. The patient was subsequently treated with 1 mg/kg of amphotericin B for a total of 14 days and recovered completely.