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Clinical and Pathophysiological Studies of Tourette Syndrome
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Clinical and Pathophysiological Studies of Tourette Syndrome

A special issue of Journal of Clinical Medicine (ISSN 2077-0383). This special issue belongs to the section "Clinical Neurology".

Deadline for manuscript submissions: closed (31 January 2022) | Viewed by 23988

Special Issue Editors

Dr. Deanna J Greene

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Guest Editor
Department of Cognitive Science, University of California San Diego, La Jolla, CA, USA
Interests: tourette syndrome; development; neurodevelopmental disorders; cognitive neuroscience; neuroimaging; brain networks; functional connectivity
Dr. Kevin Black

E-Mail Website
Guest Editor
Departments of Psychiatry, Neurology, Radiology, and Neuroscience, Washington University School of Medicine, St. Louis, MO, USA
Interests: Tourette syndrome; pharmacological neuroimaging; neuropsychiatry of movement disorders

Special Issue Information

Dear Colleagues,

Research on Tourette syndrome is growing at an ever-increasing pace. Recent insights have come from studies of natural history, epidemiology, animal models, electrophysiology, neuroimaging, pharmacology, neuropsychology, and treatments ranging from psychotherapeutic to surgical. Nevertheless, several important mysteries remain, including the following: “Why do tics tend to start at ages 5–10? Why are they more common in boys? Why do they tend to improve during sleep? Why do tics usually improve in early adulthood? How accurately can we predict outcome for an individual patient? Which patients need which treatments? Is secondary prevention possible? What is the pathophysiology associated with tics and their progression throughout development?” We invite contributions on the clinical features, treatment, or pathophysiology of Tourette syndrome and other tic disorders.

Dr. Deanna J Greene
Dr. Kevin Black
Guest Editors

Manuscript Submission Information

Manuscripts should be submitted online at www.mdpi.com by registering and logging in to this website. Once you are registered, click here to go to the submission form. Manuscripts can be submitted until the deadline. All submissions that pass pre-check are peer-reviewed. Accepted papers will be published continuously in the journal (as soon as accepted) and will be listed together on the special issue website. Research articles, review articles as well as short communications are invited. For planned papers, a title and short abstract (about 100 words) can be sent to the Editorial Office for announcement on this website.

Submitted manuscripts should not have been published previously, nor be under consideration for publication elsewhere (except conference proceedings papers). All manuscripts are thoroughly refereed through a single-blind peer-review process. A guide for authors and other relevant information for submission of manuscripts is available on the Instructions for Authors page. Journal of Clinical Medicine is an international peer-reviewed open access semimonthly journal published by MDPI.

Please visit the Instructions for Authors page before submitting a manuscript. The Article Processing Charge (APC) for publication in this open access journal is 2600 CHF (Swiss Francs). Submitted papers should be well formatted and use good English. Authors may use MDPI's English editing service prior to publication or during author revisions.

Keywords

  • Tourette syndrome
  • tic disorders
  • provisional tic disorder
  • inhibition (psychology)
  • functional connectivity
  • fMRI
  • brain/anatomy and histology
  • EEG
  • transcranial magnetic stimulation

Published Papers (6 papers)

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Research

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13 pages, 3000 KiB  
Article
Capturing Subtle Neurocognitive Differences in Children with and without Tourette Syndrome through a Fine-Grained Analysis of Design Fluency Profiles
by Mélina Tessier, Annie Desmarais, Julie B. Leclerc, Marc E. Lavoie, Kieron P. O’Connor and Bruno Gauthier
J. Clin. Med. 2022, 11(7), 1946; https://0-doi-org.brum.beds.ac.uk/10.3390/jcm11071946 - 31 Mar 2022
Cited by 3 | Viewed by 2318
Abstract
Background: Tourette syndrome (TS) can be accompanied by neurocognitive impairment. Only a few studies have focused on executive function assessment in TS using design fluency, providing preliminary results. This study aimed to characterize the detailed design fluency profile of children with TS compared [...] Read more.
Background: Tourette syndrome (TS) can be accompanied by neurocognitive impairment. Only a few studies have focused on executive function assessment in TS using design fluency, providing preliminary results. This study aimed to characterize the detailed design fluency profile of children with TS compared with neurotypical children, while addressing the central concern of frequent comorbidities in studies on TS by considering tic severity and attention-deficit/hyperactivity disorder (ADHD) symptoms and diagnosis. Methods: Sixty-one children aged between 6 and 15 years participated and were divided into a TS group (n = 28 (with ADHD n = 15)) and a control group (n = 33). Our objective was addressed by examining a wide range of measures of the Five-Point-Test, presumably sensitive to frontostriatal dysfunction. The total number of designs, repetitions, repetition ratio, unique designs, and numerical, spatial, and total strategies were examined for the total duration of the test (global measures) and at five equal time intervals (process measures). Results: The TS group produced significantly fewer numerical strategies. Groups did not differ in other global or process measures. ADHD did not affect performance. Conclusions: Children with TS do not inherently show general executive dysfunction but may present with subtle neurocognitive characteristics here revealed by comprehensive design fluency profiles. Full article
(This article belongs to the Special Issue Clinical and Pathophysiological Studies of Tourette Syndrome)
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9 pages, 767 KiB  
Article
Pain in Tourette Syndrome-Children’s and Parents’ Perspectives
by Agnieszka Małek
J. Clin. Med. 2022, 11(2), 460; https://0-doi-org.brum.beds.ac.uk/10.3390/jcm11020460 - 17 Jan 2022
Cited by 4 | Viewed by 2991
Abstract
Tourette Syndrome (TS) is a neurodevelopmental condition characterized by the presence of tics and associated behavioral problems. Yale Global Tic Severity Scale (YGTSS), The PedsQL Pediatric Pain Questionnaire, and Pediatric Pain Coping Inventory were used to assess the severity of tics, the severity [...] Read more.
Tourette Syndrome (TS) is a neurodevelopmental condition characterized by the presence of tics and associated behavioral problems. Yale Global Tic Severity Scale (YGTSS), The PedsQL Pediatric Pain Questionnaire, and Pediatric Pain Coping Inventory were used to assess the severity of tics, the severity of the pain, the location of the pain and pain coping strategies both from children’s and parents’ perspectives. Sixty percent of children demonstrated pain (past or present); the pain was reported by 72% of parents raising children with TS. The pain most commonly was cervical, throat, shoulder, ocular, and joint pain; most children declared pain located in more than one part of the body. Consistency between the declarations of children and their parents in coping with pain was observed. Pain should be recognized as a common comorbid aspect of tic disorders in childhood and therapeutic treatment must include the reduction of pain caused by tics. Full article
(This article belongs to the Special Issue Clinical and Pathophysiological Studies of Tourette Syndrome)
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12 pages, 268 KiB  
Article
Inhibitory Control in Children with Tourette Syndrome Is Impaired in Everyday Life but Intact during a Stop Signal Task
by Melanie Ritter, Signe Allerup Vangkilde, Katrine Maigaard, Anne Katrine Pagsberg, Kerstin Jessica Plessen and Julie Hagstrøm
J. Clin. Med. 2022, 11(2), 309; https://0-doi-org.brum.beds.ac.uk/10.3390/jcm11020309 - 08 Jan 2022
Cited by 1 | Viewed by 1392
Abstract
Tourette Syndrome (TS) has previously been associated with deficits in inhibitory control (IC). However, studies on IC in individuals with TS have produced conflicting results. In the present study, we investigated IC, comparing the Stop Signal Reaction Time (SSRT) measure with parent and [...] Read more.
Tourette Syndrome (TS) has previously been associated with deficits in inhibitory control (IC). However, studies on IC in individuals with TS have produced conflicting results. In the present study, we investigated IC, comparing the Stop Signal Reaction Time (SSRT) measure with parent and teacher ratings of daily life IC in 169 children aged 8–12 (60 with TS, 60 typically developing controls, 27 with attention-deficit/hyperactivity disorder (ADHD), and 22 with TS + ADHD). We further investigated associations of IC with TS and ADHD symptom severity. Children with TS showed intact SSRT performance, but impairments in daily life IC, as reported by parents and teachers. For the latter, we observed a staircase distribution of groups, with the healthy controls presenting with the best IC, followed by TS, TS + ADHD, and finally ADHD. Dimensional analyses indicated a strong association between ADHD severity and both measures of IC. Our results indicate that children with TS are not impaired in a laboratory-based measure of IC, although some difficulties were evident from measures of everyday behaviour, which may in part be due to parents and teachers interpreting tics as disinhibited behaviour. Comorbid ADHD or the severity of subthreshold ADHD symptomatology appeared to account for IC deficits. Full article
(This article belongs to the Special Issue Clinical and Pathophysiological Studies of Tourette Syndrome)
15 pages, 403 KiB  
Article
ONLINE-TICS: Internet-Delivered Behavioral Treatment for Patients with Chronic Tic Disorders
by Martina Haas, Ewgeni Jakubovski, Katja Kunert, Carolin Fremer, Nadine Buddensiek, Sebastian Häckl, Martina Lenz-Ziegenbein, Richard Musil, Veit Roessner, Alexander Münchau, Irene Neuner, Armin Koch and Kirsten Müller-Vahl
J. Clin. Med. 2022, 11(1), 250; https://0-doi-org.brum.beds.ac.uk/10.3390/jcm11010250 - 04 Jan 2022
Cited by 9 | Viewed by 2363
Abstract
Comprehensive Behavioral Intervention for Tics (CBIT) is considered a first-line therapy for tics. However, availability of CBIT is extremely limited due to a lack of qualified therapists. This study is a multicenter (n = 5), randomized, controlled, observer-blind trial including 161 adult [...] Read more.
Comprehensive Behavioral Intervention for Tics (CBIT) is considered a first-line therapy for tics. However, availability of CBIT is extremely limited due to a lack of qualified therapists. This study is a multicenter (n = 5), randomized, controlled, observer-blind trial including 161 adult patients with chronic tic disorders (CTD) to provide data on efficacy and safety of an internet-delivered, completely therapist-independent CBIT intervention (iCBIT Minddistrict®) in the treatment of tics compared to placebo and face-to-face (f2f) CBIT. Using a linear mixed model with the change to baseline of Yale Global Tic Severity Scale-Total Tic Score (YGTSS-TTS) as a dependent variable, we found a clear trend towards significance for superiority of iCBIT (n = 67) over placebo (n = 70) (−1.28 (−2.58; 0.01); p = 0.053). In addition, the difference in tic reduction between iCBIT and placebo increased, resulting in a significant difference 3 (−2.25 (−3.75; −0.75), p = 0.003) and 6 months (−2.71 (−4.27; −1.16), p < 0.001) after the end of treatment. Key secondary analysis indicated non-inferiority of iCBIT in comparison to f2f CBIT (n = 24). No safety signals were detected. Although the primary endpoint was narrowly missed, it is strongly suggested that iCBIT is superior compared to placebo. Remarkably, treatment effects of iCBIT even increased over time. Full article
(This article belongs to the Special Issue Clinical and Pathophysiological Studies of Tourette Syndrome)
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13 pages, 419 KiB  
Article
Acute and Long-Term Effects of an Internet-Based, Self-Help Comprehensive Behavioral Intervention for Children and Teens with Tic Disorders with Comorbid Attention Deficit Hyperactivity Disorder, or Obsessive Compulsive Disorder: A Reanalysis of Data from a Randomized Controlled Trial
by Lilach Rachamim, Hila Mualem-Taylor, Osnat Rachamim, Michael Rotstein and Sharon Zimmerman-Brenner
J. Clin. Med. 2022, 11(1), 45; https://0-doi-org.brum.beds.ac.uk/10.3390/jcm11010045 - 23 Dec 2021
Cited by 4 | Viewed by 2389
Abstract
Attention deficit hyperactivity disorder (ADHD), obsessive compulsive disorder (OCD) and tic disorders (TD) commonly co-occur. In addition, specific inattention difficulties and poor impulse control are related to TD in the absence of comorbid ADHD. In this study we reanalyzed data from a recently [...] Read more.
Attention deficit hyperactivity disorder (ADHD), obsessive compulsive disorder (OCD) and tic disorders (TD) commonly co-occur. In addition, specific inattention difficulties and poor impulse control are related to TD in the absence of comorbid ADHD. In this study we reanalyzed data from a recently completed study comparing internet-delivered, self-help comprehensive behavioral intervention for tics (ICBIT) with a waiting-list control group. The current study describes the effects of an (ICBIT) in children and adolescents with TD with and without comorbid diagnoses of ADHD or OCD at post intervention and over three- and six-month follow-up periods. Thirty-eight 7 to 18-year-olds completed the ICBIT. Of these, 16 were diagnosed with comorbid ADHD and 11 were diagnosed with OCD. A significant improvement in tic measures was found in all groups. Both the TD + ADHD and the TD − ADHD groups were similar in the magnitude of tic reduction from baseline to post-treatment, and at the three and six-month follow-up assessments. However, the TD + OCD group benefitted less from intervention than the TD—OCD group. There were meaningful reductions in parental reports of inattention, as well as hyperactive and impulsive symptoms at post intervention and over the 6-month follow-up period. Thus, ICBIT can be effectively delivered in the presence of comorbid ADHD or OCD symptomatology and may reduce symptoms of inattention and impulsivity. Larger studies of ICBIT in children and teens with TD and comorbid ADHD and OCD are needed to optimize responses to ICBIT. Full article
(This article belongs to the Special Issue Clinical and Pathophysiological Studies of Tourette Syndrome)
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Review

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32 pages, 542 KiB  
Review
A Comprehensive Review of Tic Disorders in Children
by Keisuke Ueda and Kevin J. Black
J. Clin. Med. 2021, 10(11), 2479; https://0-doi-org.brum.beds.ac.uk/10.3390/jcm10112479 - 03 Jun 2021
Cited by 35 | Viewed by 10816
Abstract
Tics are characterized by sudden, rapid, recurrent, nonrhythmic movement or vocalization, and are the most common movement disorders in children. Their onset is usually in childhood and tics often will diminish within one year. However, some of the tics can persist and cause [...] Read more.
Tics are characterized by sudden, rapid, recurrent, nonrhythmic movement or vocalization, and are the most common movement disorders in children. Their onset is usually in childhood and tics often will diminish within one year. However, some of the tics can persist and cause various problems such as social embarrassment, physical discomfort, or emotional impairments, which could interfere with daily activities and school performance. Furthermore, tic disorders are frequently associated with comorbid neuropsychiatric symptoms, which can become more problematic than tic symptoms. Unfortunately, misunderstanding and misconceptions of tic disorders still exist among the general population. Understanding tic disorders and their comorbidities is important to deliver appropriate care to patients with tics. Several studies have been conducted to elucidate the clinical course, epidemiology, and pathophysiology of tics, but they are still not well understood. This article aims to provide an overview about tics and tic disorders, and recent findings on tic disorders including history, definition, diagnosis, epidemiology, etiology, diagnostic approach, comorbidities, treatment and management, and differential diagnosis. Full article
(This article belongs to the Special Issue Clinical and Pathophysiological Studies of Tourette Syndrome)
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