The authors describe a rare case of a pediatric patient with focal inhibitory motor seizures (ictal paralysis) with the seizure focus in the primary motor cortex. Seizures were characterized by ictal paralysis of the contralateral limbs without loss of consciousness. The seizure focus and progression were examined using long-term video electroencephalography (EEG) monitoring and MR imaging. Invasive recordings of the ictal EEG revealed spike rhythms arising from the primary motor cortex, suggesting that seizures associated with paralysis can arise from hyperactivity in either positive or negative motor areas. Resection of the cortical area related to ictal onset resulted in a seizure-free status for the last 3 years.