Surgical treatment of intractable epilepsy presenting with hyperkinetic seizures originating in the frontal lobe

Hyperkinetic seizures (HKS) are characterized by complex movements involving the proximal segment of the limbs and trunk. The proximal predominance of these movements results in large movements, thus the label“hyperkinetic.”Previous reports suggested the orbitofrontal and anterior cingulate cortex are associated with HKS. We describe the two male patients with frontal lobe epilepsy who manifest HKS. Both were successfully treated by surgical resection. The patients experienced their first seizures at eleven years old and five years old. Both cases exhibited progressive, intractable HKSs and intellectual deterioration. In both, HKS was misdiagnosed as a psychosis. At the ages of nineteen, they were evaluated by a comprehensive epilepsy monitoring unit. FLAIR MR images revealed thickened gray matter and hyperintensity of white matter at right anterior cingulate cortex in one case and that at left frontal lobe involving orbitofrontal area in the other case. Long‒term video electroencephalography (EEG) showed interictal spikes in fronto‒temporal area in both cases. Ictal EEG was non‒localizable in the former, and attenuation in left frontal area in the latter. The former was evaluated using intracranial EEG recording, and revealed that the anterior cingulate cortex was the seizure‒onset zone. Followed focal cortical resection was performed. The latter was done the extended frontal lobectomy with preservation of the language area since functional mapping is not ideal in patients with mental retardation. Both patients emerged seizure‒free. This work underscores the importance of considering HKS, which is easily misdiagnosed as a psychiatric disorder, as a manifestation of frontal lobe epilepsy.