The impact of epileptiform activity on the functional organization of various cortical regions is currently a widely discussed topic. Our patient, suffering from left-sided temporal lobe epilepsy with typical mesiotemporal sclerosis, had a remarkable history of a clear-cut handedness shift in early childhood. In this study, we considered the possibility of the functional reorganization of the motor cortex using functional MRI. The results reflect epileptiform activity as an important formative factor for motor cortex development in our patient.