Tourette syndrome (TS) is a common childhood onset neuropsychiatric disorder, affecting approximately 1% of children. Tourette Syndrome is defined by multiple motor and one or more vocal tics (repeated, largely involuntary movements/noises) that endure for more than a year (APA 2000). Here we report a child patient with severe TS, who had significant improvement in social functioning with pimozide treatment. The case is a 12-year-old male, with 4 years old TS. Disease onset occurred in 2009 with involuntary, repetitive muscle movements and vocalizations. No triggering factors or accompanying symptoms could be identified. Family history was negative for sleep or other neurologic disorders. Comorbidities, obsessive-compulsive disorder have been diagnosed by clinical interview and attention deficit/hyperactivity disorder was excluded by clinical interview. A routine electroencephalogram revealed no abnormalities and indicated the absence of epileptiform activities. He was treated with risperidone (1.5 mg/day), haloperidol (5 mg/day) in sequence between 2009 and 2012. These drugs produced either mild improvement or no effect. In addition, he had an elevated prolactin level after taking risperidone. We initiated pimozide at 3 mg/d, şuoxetine 20 mg/d and continued treatment for 6 consecutive months. Six weeks after the initiation of treatment, improvement in both the involuntary muscle movements and the vocalizations was noticed (on the Yale Global Tic Severity Scale, the total motor score decreased from 24 to 13 and the total phonic score decreased from 12 to 0). The patient stated that he experienced a significant improvement of the symptoms. Objective: The beneficial effects of pimozide on both motor and vocal tics are shown in this report and in previous studies. The Cochrane review of Pringsheim and Marras (2009) included six randomized controlled trials on pimozide in TS. In total, 162 people were included with an age-range of 7-53 years. Pimozide was compared with risperidone (two trials), haloperidol (one trial), haloperidol and placebo (two trials), and placebo (one trial). The two studies that compared pimozide and risperidone reported no significant differences in terms of tic reduction or adverse reactions. In contrast to haloperidol, pimozide had fewer adverse reactions but was also slightly less effective. Additionally, the studies revealed that pimozide was more effective than placebo. Pimozide appears relatively safe for use in pediatric TS. This finding will help guide medication selection in child patients with specific medical vulnerabilities.