In children with benign childhood epilepsy with centrotemporal spikes, centrotemporal spikes may cause language dysfunction via disruption of underlying functional neuroanatomy. Fifteen patients with benign childhood epilepsy with centrotemporal spikes and 15 healthy controls completed 3 functional magnetic resonance imaging (MRI) language paradigms; standardized cognitive and language assessments were also performed. For all paradigms, children with benign childhood epilepsy with centrotemporal spikes showed specific regional differences in activation compared to controls. Children with benign childhood epilepsy with centrotemporal spikes also differed from controls on neuropsychological testing. They did not differ in general intelligence, but children with benign childhood epilepsy with centrotemporal spikes scored significantly lower than controls on tests of language, visuomotor integration, and processing speed. These results extend previous findings of lower language and cognitive skills in patients with benign childhood epilepsy with centrotemporal spikes, and suggest epilepsy-related remodeling of language networks that may underlie these observed differences.