Controversy exists over the pathophysiology of Tourette syndrome (TS). The case reported is a 37-year-old unmarried man suffering from both TS and complex partial epilepsy (CPE). He began to have seizures at 2-3 months of age. The CPE featured dark vision, dizziness, followed by unresponsiveness, a blank stare, occasional loss of posture control, and occasional automatism consisting of going to the toilet to urinate. TS gradually began to develop when he was 3-4 years of age. The tics were characterized by stereotypic stuttering, vocalization, hiccups, grimacing, snorting, and jerky supination of both forearms. EEG sharp waves with phase reversal at the left frontotemporal region were present but they were not related to the tics. Magnetic resonance imaging revealed atrophy of the left temporal and frontal lobes, as well as absence of normal asymmetry of basal ganglia. This case supports the theory that TS is related to the left frontal lobe, limbic system, and basal ganglia, but contradicts the hypothesis that the tics are ictal events.