We report a 27-year-old man with pilomotor seizures originating in the right frontal lobe. Subtracted ictal SPECT coregistered with MRI showed multifocal hyperperfused areas in the anterior medial frontal area, mainly involving the cingulate gyrus. Chronic electrocorticography with subdural electrode arrays during the piloerection demonstrated that the pilomotor seizures were originating in the anterior medial frontal region. After resection of the focus, the patient became seizure free.