Introduction. A sudden withdrawal of antiepileptic drugs gives higher rate of epileptic seizures in the settings of video electroencephalography (vEEG), monitoring that is a subject to further registration and analysis. A very rare complication of this method is transient lesion of the splenium of corpus callosum (SCC) detected with brain MRI. Case report. We presented a patient with a 5-year history of pharmacoresistant epilepsy admitted to the Institute of Neurology (August, 2008) for vEEG monitoring. Interictal epileptic discharges but none seizure were recorded after the sudden withdrawal of antiepileptic medications, during 5 days of vEEG monitoring Initial brain MRI three days after vEEG monitoring revealed focal lesion in SCC, hyperintense on T2 and FLAIR sequence. A longitudinal radiological follow-up (7 and 49 days after initial acquisition) confirmed transient nature of the lesion without diffusion coefficient changes. Conclusion. SCC lesion, reversible and harmless, may occur after a sudden antiepileptic withdrawal. Its early detection makes further extensive neuroradiological and clinical examinations unnecessary.