| Summary: | Malaria-related hemophagocytic lymphohistiocytosis is a rare, potentially fatal, hyperinflammatory disease entity which can be challenging to diagnose and treat. It is usually associated with Plasmodium falciparum infection. It is less frequently associated with Plasmodium vivax. Here we report an unusual case of a 23-year-old healthy Nigerian man who presented with fever, microangiopathic hemolytic anemia, acute renal failure, and confusion, and was diagnosed as having cerebral malaria-related hemophagocytic lymphohistiocytosis caused by P. vivax infection. He was successfully treated with intravenous artesunate and doxycycline with dramatic clinical improvement. Keywords: Plasmodium vivax, Malaria, Hemophagocytic lymphohistiocytosis, Artesunate, Doxycycline
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