A developmentally normal 4-year-old white female who presented with pa
in in the right hand as the only manifestation of epilepsy is reported
. Two years later, she developed complex partial seizures following ri
ght-hand pain. Computed tomography and magnetic resonance imaging were
unremarkable. Prolonged ambulatory electroencephalography (EEG) as we
ll as video-EEGs with ictal pain episodes failed to reveal abnormaliti
es. Only a full night video-EEG performed after antiepileptic drug wit
hdrawal demonstrated 2 right-hand pain episodes followed by a complex
partial seizure with ictal epileptiform activity on the scalp EEG in t
he left parasagittal area, rapidly generalized and interictal discharg
es in the C3-P3 area. This patient had a very unusual presentation of
epilepsy.