Acute hemorrhagic leukoencephalitis (AHLE, Hurst's disease) is a rare hyperacute variant of acute disseminated encephalomyelitis (ADEM) characterized by severe, rapidly progressive clinical illness and hemorrhagic necrosis of white matter. Like ADEM, it is often preceded by viral illness or vaccination. Plasmodium vivax infection is usually uncomplicated and non-fatal with only a handful of reports of central nervous system complications. In this article, we report a previously unknown association between AHLE and P. vivax infection.