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A 3-year-old female presented with splenomegaly and hypersplenism manifestations, including anemia, thrombocytopenia, and abdominal pain/distention. All common metabolic, hematologic, gastrointestinal, and infectious etiologies for splenomegaly were excluded. Diagnosis of idiopathic splenic peliosis was made and splenectomy was recommended. History revealed that the patient's mother had acquired a nonspecific skin infection during a visit to the Middle East 2 years prior. Serum antibodies and confirmatory PCR testing for visceral leishmaniasis were positive. After treatment with liposomal amphotericin, at 6-month follow-up her hypersplenism manifestations had resolved and her splenomegaly had significantly decreased. Visceral leishmaniasis should be considered in cases of marked splenomegaly, anemia, and thrombocytopenia, especially with a history of visiting an endemic area.

Visceral Leishmania as Unusual Cause of Splenic Peliosis in the United States