Enterovirus infection has been rarely reported to cause cerebral infarction in infants. We describe a 2-month-old boy with right focal seizure and right hemiparesis associated with enterovirus infection during an epidemic of enterovirus 71 infection in Taiwan in 1998. Magnetic resonance imaging and angiography showed vasculitis in the left anterior cerebral artery with cerebral infarction. In the unclarified pathogenesis of cerebral disease in enterovirus infection, this case suggests focal vasculitis with subsequent cerebral infarction. Enterovirus-related vasculitis of the central nervous system is thus another consideration when facing a child with focal seizure, acute hemiplegia and cerebral infarction.