We report the first case of a pilomotor seizure detected by electroencephalography /functional magnetic resonance imaging (EEG/fMRI). An adult woman presented with history of bouts of gooseflesh feeling and poilomotor activity in the left leg following viral encephalitis. 24-hour video-EEG and simultaneous EEG during fMRI revealed ictal discharges in the right parietal and temporal lobes. Associated blood oxygen level- dependent (BOLD) activations were found mainly in the right parietal region. The result represents a different generator of pilomotor seizure compared to prior reports. We suggests that the feeling of gooseflesh could be the core ictal symptom and a direct pathway from the sensory cortex to the lower autonomic system may exist bypassing the classic cerebral autonomic center.