Musicogenic epilepsy (ME) is a condition in which seizures are triggered by music. We describe a novel investigation of the mechanisms of ME using noninvasive (ictal 2-flurodeoxyglucose (2-FDG) positron emission tomography (PET)) and invasive (subdural arrays and depth electrodes) methodology in one patient with ME. Ictal PET defined onsets in the right mesial temporal lobe. This was confirmed by intracranial electrode recordings, where there was onset in the right mesial temporal lobe, followed by sequential spread to the lateral temporal cortex, Heschel's gyrus, insula, and frontal lobes. Right temporal lobectomy resulted in Engel class 1 outcome.