Hemophagocytic lymphohistiocytosis (HLH) is a rare debilitating condition that can be triggered by an infectious cause, often linked to the Epstein-Barr virus (EBV). In this case, we evaluated a patient with pancytopenia. The bone marrow aspiration revealed the presence of amastigotes and active hemophagocytosis, indicating that the HLH was induced by a
Leishmania
infection. The patient was treated with lyophilized amphotericin B followed by miltefosine, which effectively resolved the infection and HLH. This case report underscores the presentation and findings of
Leishmania
-induced HLH, as well as the successful treatment approach that led to improved patient outcomes.