Case report of chapeau de gendarme or ictal pouting seizure of insular origin in a teenager

Summary We report a teenager with childhood onset ‘chapeau de gendarme’ or ‘ictal pouting’ seizures of anterior insular lobe origin. Chapeau de gendarme ictal sign has been associated with frontal lobe epilepsy, however in this case, we confirmed with stereo-electroencephalography (SEEG) that electrical seizures were starting from the anterior insular cortex prior to her typical clinical manifestations. Surgical resection of the insular and frontal-lobe network resulted in seizure freedom. We propose that the anterior insular cortex should be included during phase 2 investigations of chapeau de gendarme or ictal pouting seizures.