Brain magnetic resonance imaging of an 11-year-old male with Tourette syndrome demonstrated multicystic changes predominately in the gyrus rectus of the left frontal lobe. Other brain regions, including the basal ganglia, were normal. He did not have any symptoms of the comorbid conditions associated with Tourette syndrome, such as attention-deficit disorder or obsessive-compulsive disorder. The possible neurobiologic connection between Tourette syndrome and the gyrus rectus and its interconnecting pathways is discussed.