Abstract

Some individuals with Tourette syndrome (TS) have severe motoric and vocal tics that interfere with all aspects of their lives, while others have mild tics that pose few problems. We hypothesize that observed tic severity reflects a combination of factors, including the degree to which dopaminergic (DA) and/or noradrenergic (NE) neurotransmitter systems have been affected by the disorder, and the degree to which the child can exert cognitive control to suppress unwanted tics. To explore these hypotheses, we collected behavioral and eyetracking data from 26 patients with TS and 26 controls between ages 7 and 14, both at rest and while they performed a test of cognitive control. To our knowledge, this is the first study to use eyetracking measures in patients with TS. We measured spontaneous eyeblink rate as well as pupil diameter, which have been linked, respectively, to DA and NE levels in the central nervous system. Here, we report a number of key findings that held when we restricted analyses to unmedicated patients. First, patients’ accuracy on our test of cognitive control accounted for fully 50% of the variance in parentally reported tic severity. Second, patients exhibited elevated spontaneous eyeblink rates compared to controls, both during task performance and at rest, consistent with heightened DA transmission. Third, although neither task-evoked pupil dilation nor resting pupil diameter differed between TS patients and controls, pupil diameter was positively related to parentally reported anxiety levels in patients, suggesting heightened NE transmission in patients with comorbid anxiety. Thus, with the behavioral and eyetracking data gathered from a single task, we can gather objective data that are related both to tic severity and anxiety levels in pediatric patients with TS, and that likely reflect patients’ underlying neurochemical disturbances.

Highlights

  • Tourette Syndrome (TS) is a neurodevelopmental disorder characterized by non-purposeful, rapid, recurrent vocalizations, and movements, frequently accompanied by unusual and uncomfortable sensations or urges [1,2,3]

  • Significant group differences were observed regardless of which set of patients was considered for full-scale IQ [Wechsler Abbreviated Scale of Intelligence (WASI)], behavioral self-regulation (BRIEF), child behavior problems (CBCL), attention-deficit hyperactivity disorder (ADHD) symptoms (Conner’s), obsessive–compulsive symptoms (CY-BOCS), and tic severity (YGTSS)

  • Patients exhibited a trend toward higher anxiety levels (MASC Anxiety Index) than controls

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Summary

Introduction

Tourette Syndrome (TS) is a neurodevelopmental disorder characterized by non-purposeful, rapid, recurrent vocalizations, and movements, frequently accompanied by unusual and uncomfortable sensations or urges [1,2,3]. A diagnosis of “pure” TS is the exception rather than the rule; over 80% of TS patients have been diagnosed with comorbid mental health conditions, most frequently attention-deficit hyperactivity disorder (ADHD) and obsessive–compulsive disorder (OCD) [4,5,6]. ADHD, and OCD have all been characterized as disorders implicating frontostriatal circuitry [7,8,9,10], which underpins both motoric and cognitive control [11]. While altered motor control is a defining characteristic of TS, there is mixed evidence of altered cognitive control in this disorder [11]. We tested whether behavioral measures of cognitive control, along with spontaneous eyeblink rate and/or pupil diameter measured at rest and during execution of a cognitive control task, could help to differentiate patients with TS from one another and/or from controls

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