Introduction: Gilles de la Tourette syndrome is characterized by involuntary vocal and/or motor tics. It affects 1-2% of school children and is the most common movement disorder in pediatric group (1). Spontaneous remission rate is high, and treatment is required for those who failed to improve and experience social problems (2).

Here a 11 year-old girl, who failed different treatments and refused going to school due to tics but showed a dramatic improvement with topiramate, is presented.

Case: She has complaints of bending her neck and body to right side, and making a high-pitched cry for four years, and  used, respectively, risperidone (1 year), risperidone + fluoxetine (6 months), pimozide (3 months) and amisulperid (3 months). When she applied with symptoms, she used no medicine, had normal EEG and brain MRI. Second EEG was also normal. Topiramate was initiated, her complaints resolved from the second week on (2 mg/kg/day), and remained tics-free during 5 months follow-up period.

Discussion: The pathophysiology of TS is not clearly understood yet. The most common therapeutic agents include typical narcoleptics (haloperidol, pimozide), atypical narcoleptics (risperidone, aripiprazole, quetiapine, ziprasidone), alpha-adrenergic agonists (clonidine, guanfacine), benzamides (sulpiride, tiapride), selective noradrenaline reuptake inhibitors (atomoxetine) and oxcarbazepine (2). An optimum treatment guideline could not be established. Controlled studies are not sufficient in the literature. Recommendations include specialists’ experiences and preferences (2). Topiramate was reported to reduce tics in some patients (3,4).

This case is valuable to highlight topiramate as the treatment option in TS patients who failed conventional pharmacotherapy.