A 33-year-old male patient with fever, splenomegaly, pancytopenia and lymphocytosis was admitted to the Department of Hematology in Rijeka. Laboratory findings, bone marrow aspiration and biopsy excluded hemoblastosis and aplastic anemia. To exclude primary splenic lymphoma we performed splenic aspiration where Leishmania amastigotes were found. No cases of visceral leishmaniasis have been previously described in the Northern Adriatic region. Considering epidemiology, a contraction of the disease in the Velebit mountain range could be possible despite the current non-endemic status of the region.