West Nile virus (WNV) is a neurotropic flavivirus and the leading cause of mosquito-borne diseases in North America. In approximately 20% of cases, infection leads to a self-limited febrile illness and, in less than 1%, to a neuroinvasive disease often manifesting as meningoencephalitis with or without acute flaccid paralysis.1 Herein, we report the first case of WNV-associated CNS vasculitis and intracranial hemorrhage, thus expanding the spectrum of WNV infection.