Visseral leishmaniasis is a protozoan infection that is characterized by a prolonged fever, splenomegaly, hepatomegaly, hypergammaglobulinemia, progressive anemia, and pancytopenia. Splenic infarct is a rare problem, which is seen over the course of this disease. In this study, a four-year-old female patient diagnosed with visceral leishmaniasis who developed resistance to sodium stibogluconate as well as a splenic infarct is presented. Abdominal ultrasonography and computerized tomography, which were used because of the development of left upper abdominal pain on the third day of the treatment, revealed a splenic infarct. Despite the clinical improvement at the end of the first week, enlargement in the liver and the spleen along with high fever were reported on the twentieth day of the treatment. Because of the resistance to pentavalent antimonial drug, amphotericin B liposomal treatment was administered and a positive clinical response was observed. Following the treatment of only the primary disease, the splenic infarct resolved at the end of the first month without any complications. This case is presented as both drug resistance and splenic infarct are rarely seen during the course of visceral leishmaniasis

Keywords: Splenic infarction, Drug resistance, Leishmaniasis, Visceral