Deep brain stimulation is invasive and used in selected patients with intractable Tourette Syndrome. The anaesthetic technique of first choice during implantation of the electrodes is an awake technique with local anaesthetics and conscious sedation. The anaesthetic management can be challenging, especially in Tourette Syndrome patients due to sudden movements. The anaesthetic goal is to have a comfortable patient during the procedure with minimal influence of pharmacological agents on microelectrode recordings. Here, we describe our experience with the use of an intravenous dexmedetomidine infusion during deep brain stimulation in a patient with Tourette Syndrome. We discuss the effects on the microelectrode recordings and clinical testing. Finally, we describe the advantages of dexmedetomidine as a sedative agent in Tourette Syndrome patients during deep brain stimulation and the pharmacological effects of dexmedetomidine on the basal ganglia.