Visceral leishmaniasis, hemophagocytic lymphohistiocytosis, and intracerebral hemorrhage – a concerning triad

Background: Visceral leishmaniasis (VL) is a rare cause of hemophagocytic lymphohistiocytosis (HLH), a severe hyperinflammatory syndrome. The development of intracerebral hemorrhage (ICH) in this context is exceedingly rare, adding a unique and life-threatening dimension to this triad. Case Presentation: A middle-aged man presented with a 4-month history of recurrent fever. He also reported progressive breathlessness, a sensation of heaviness in the left abdomen, and pedal edema over the past 4 weeks. Additionally, he experienced black discoloration of urine and stool for 2 weeks. Physical examination revealed massive splenomegaly, melena, hematuria, and pancytopenia. Bone marrow aspiration confirmed hemophagocytosis, and the rk39 test for VL was positive. An H-score of 234 indicated a 98-99% probability of HLH. The patient was initiated on treatment with a single dose of liposomal amphotericin-B. However, on the fourth day of therapy, he developed a sudden onset of headache followed by altered sensorium. Neuroimaging revealed ICH with surrounding edema and intraventricular extension, causing a significant mass effect. He was managed conservatively with three units of single-donor platelets and showed gradual neurological improvement without further invasive intervention. He was eventually discharged in a hemodynamically stable condition. Conclusion: This case highlights the potential coexistence of VL, HLH, and ICH, a triad that poses significant diagnostic and therapeutic challenges. Early recognition and tailored management are crucial for favorable outcomes in such rare and complex presentations.