BACKGROUND: The benefits of neurosurgery in Tourette Syndrome (TS) are still incompletely understood. Prefrontal cortical electrical stimulation offers a less invasive alternative to deep brain stimulation. OBJECTIVE: To perform a pilot assessment on safety and efficacy of prefrontal cortical bilateral electrical stimulation in TS using clinical and brain metabolic assessments. METHODS: Four adult TS patients underwent tic assessment using the Yale Global Tic Severity Scale and the Rush Video Rating Scale at baseline and 1, 3, 6, and 12-months after implant; whereas FDG-PET scans were acquired at baseline and after 6 and 12 months. RESULTS: Tic clinical scores were improved at 6 months after implant, meanwhile they showed a tendency to re-emerge at the 12-month follow-up. There was a correlation between FDG-PET and tics, mainly consisting in a reduction of baseline brain hypermetabolism, which paralleled tic score reduction. CONCLUSION: Epidural stimulation in TS is safe and yields a modulation of tics, paralleled by FDG-PET metabolic modulation.