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Tourette's syndrome is a neuropsychiatric disorder characterized by formidable motor and vocal tics. Many individuals also present with comorbid neuropsychiatric conditions. Though patients often benefit from pharmacological and behavioral therapies, a subset of individuals develop severe, treatment-resistant symptoms that might necessitate more invasive interventions, such as Deep Brain Stimulation (DBS). DBS, particularly targeting regions like the globus pallidus internus (GPi) and the centromedian-parafascicular complex (CM-Pf) of the thalamus, has demonstrated effectiveness in reducing tic severity and improving quality of life. This review outlines the mechanism, clinical efficacy, and long-term outcome of DBS in TS. Results from clinical studies reveal significant reductions in tics. However, success with DBS is variable depending on a number of factors, including target selection and electrode placement. The use of DBS has ethical considerations, which include risks to the surgical procedure, the need for full and complete informed consent, and questions about the implications of such treatment on cognitive and emotional growth. Long-term follow-up will be required to ensure appropriate patient outcomes and complication management. Additional research and ethical debate will be needed with advancing DBS technology to ensure responsible and equitable treatment. This paper narratively summarizes the surgical options available for TS, with a focus on the current status of DBS in the management of the disease. [ABSTRACT FROM AUTHOR]

Background: The occurrence of tics is the main basis for the diagnosis of Gilles de la Tourette syndrome (GTS). Video‐based tic assessments are time consuming. Objective: The aim was to assess the potential of automated video‐based tic detection for discriminating between videos of adults with GTS and healthy control (HC) participants. Methods: The quantity and temporal structure of automatically detected tics/extra movements in videos from adults with GTS (107 videos from 42 participants) and matched HCs were used to classify videos using cross‐validated logistic regression. Results: Videos were classified with high accuracy both from the quantity of tics (balanced accuracy of 87.9%) and the number of tic clusters (90.2%). Logistic regression prediction probability provides a graded measure of diagnostic confidence. Expert review of about 25% of lower‐confidence predictions could ensure an overall classification accuracy above 95%. Conclusions: Automated video‐based methods have a great potential to support quantitative assessment and clinical decision‐making in tic disorders. [ABSTRACT FROM AUTHOR]

This is a protocol for a Cochrane Review (prototype). The objectives are as follows: This systematic review aims to summarize the available scientific literature on the effect of mindfulness‐based approaches in children and adults with Chronic Tic Disorder (CTD) or Tourette Disorder (TD). More specifically, the research question is: What is the effectiveness of mindfulness‐based interventions in reducing tic severity and improving overall functioning, quality of life, and co‐occurring symptoms in children and adults with CTD or TD? [ABSTRACT FROM AUTHOR]

Tic disorders in children often co-occur with other disorders that can significantly impact functioning. Screening for quality of life (QoL) can help identify optimal treatment paths. This quality improvement (QI) study describes implementation of a QoL measure in a busy neurology clinic to help guide psychological intervention for patients with tics. Using QI methodology outlined by the Institute for Healthcare Improvement, this study implemented the PedsQL Generic Core (4.0) in an outpatient medical clinic specializing in the diagnosis and treatment of tic disorders. Assembling a research team to design process maps and key driver diagrams helped identify gaps in the screening process. Conducting several plan-do-study-act cycles refined identification of patients appropriate to receive the measure. Over the three-year study, electronic health record notification tools and data collection were increasingly utilized to capture patients' information during their visit. Over 350 unique patients were screened during the assessment period. Electronic means replaced paper measures as time progressed. The percentage of patients completing the measure increased from 0% to 51.9% after the initial implementation of process improvement, advancing to 91.6% after the introduction of electronic measures. This average completion rate was sustained for 15 months. Using QI methodology helped identify the pragmatics of implementing a QoL assessment to enhance screening practices in a busy medical clinic. Assessment review at the time of appointment helped inform treatment and referral decisions. [ABSTRACT FROM AUTHOR]

Introduction: Persistent Tic Disorders such as Tourette Syndrome are common neurodevelopmental disorders that are highly stigmatized. Many individuals with Persistent Tic Disorders experience peer rejection, loneliness, and self-stigma. Experiencing stigmatization during childhood can influence the persistence of moderate-to-severe tics later in life. Additionally, these factors have been associated with increased suicidal ideation, suicide attempts, and psychiatric symptom severity. There is a need for interventions to reduce stigma and stigmatization in Persistent Tic Disorders. Before developing cost-effective interventions to mitigate stigma's profound downstream health impacts, a reliable measure of stigmatization must be created. The overarching goal of this research is to develop and validate the Tourette Discrimination-Stigmatization (TD-STIGMA) Scale. Methods: This paper presents the study protocol for developing and validating the TD-STIGMA Scale. The study is designed as a mixed methods study to develop the TD-STIGMA scale and evaluate its psychometric properties. The study uses a phased approach: (1) collection of narrative and thematic content data through in-depth qualitative interviews of stakeholders, (2) development of a novel TD-STIGMA self-report scale using the Delphi Method based on these results, and (3) completion of analyses to determine the scale's psychometric properties (confirmatory factor analysis, convergent, known-group, criterion validity, and test-retest reliability). Discussion: This project will result in a personalized approach to stigma measurement about youth and young adults with Persistent Tic Disorders, which to date does not exist. There are several limitations. Comorbidities or spiritual or cultural beliefs may affect perceptions of stigma and are not directly assessed in this study. We will utilize institutional resources for community outreach to purposefully sample underrepresented minorities who may be at disproportionate risk of adverse outcomes. However, this may not be fully representative of the generalized tic population. The study team will be purposeful in maintaining participant engagement for study retention. Lastly, participants from a tertiary referral center may not fully represent the generalized tic community. However, we hope our broad recruitment strategy and virtual study visits will facilitate a diverse and inclusive sampling of the patient population. [ABSTRACT FROM AUTHOR]

• A patient-centered approach was implemented to define comprehensive behavioral intervention for tics (CBIT) research priorities. • Priorities related to treatment access and improving broad functional outcomes. • Our approach efficiently identified actionable priorities for CBIT research. To establish a patient-centered agenda for research that will lead to effective, widespread availability, adoption, and utilization of evidence-based behavioral treatment of Tourette syndrome and other tic disorders (TDs), we planned and executed a multistage, collaborative "Treating Tourette Together" research planning project with researchers, clinicians, patients, families, and other interested parties. Priorities for future behavioral treatment research were solicited from these parties via anonymous community surveys, a 2-day research planning summit with 46 individuals representing key stakeholder groups, and community response to summit reports. Four high-priority research domains were identified: (a) expanding treatment access, (b) improving treatment outcomes, (c) optimizing treatment within a broader care model, and (d) evaluating outcomes beyond tic severity. Community-engaged participatory research models can efficiently delineate clear and actionable priorities for clinical research. This approach holds promise for improving the impact of clinical research in TDs and other neuropsychiatric disorders. [ABSTRACT FROM AUTHOR]

In this, the tenth annual update for the F1000Research Tics collection, we summarize research reports from 2023 on Gilles de la Tourette syndrome and other tic disorders. The authors welcome article suggestions and thoughtful feedback from readers.

Limited data are available regarding the impact of the coronavirus disease 2019 (COVID-19) pandemic on adolescents with Tourette syndrome (TS). We sought to compare sex differences in tic severity experienced by adolescents before and during the COVID-19 pandemic. We extracted from the electronic health record and retrospectively reviewed Yale Global Tic Severity Scores (YGTSS) from adolescents (ages 13 through 17) with TS presenting to our clinic before (36 months) and during (24 months) the pandemic. A total of 373 unique adolescent patient encounters (prepandemic: 199; pandemic: 173) were identified. Compared with prepandemic, girls accounted for a significantly greater proportion of visits during the pandemic (p < 0.001). Prepandemic, tic severity did not differ between girls and boys. During the pandemic, compared with girls, boys had less clinically severe tics (p = 0.003). During the pandemic, older girls, but not boys, had less clinically severe tics (ρ =− 0.32, p = 0.003). These findings provide evidence that, regarding tic severity assessed with YGTSS, the experiences of adolescent girls and boys with TS have differed during the pandemic. [ABSTRACT FROM AUTHOR]

Objective: To review the current state of the literature regarding anxiety symptoms and anxiety disorders in chronic tic disorder (CTD). Results: We conducted a literature search on anxiety and tic disorders. Anxiety symptoms and anxiety disorders are common in youth with CTD, with ∼30%–50% of youth with CTD having at least one co-occurring anxiety disorder. Tics often improve by young adulthood but anxiety symptoms tend to persist, or worsen, over time. Anxiety and tics are closely related, but the exact nature of their relationship is poorly understood. We discuss some potential ways in which anxiety and tics are linked with an emphasis on the underlying brain circuitry involved. The relationship between anxiety and tics may be related to the premonitory urge. In addition, stress hormones may link anxiety and tics. Individuals with CTD have greater activation of their hypothalamic-pituitary-adrenal system in response to acute stress. We also review the impact of anxiety on youth with CTD and approaches to management of anxiety in youth. Conclusions: Anxiety is common in youth with CTD, is associated with more severe CTD, and can adversely affect a child's function. Thus, it is important to identify anxiety disorders in CTD and manage them appropriately. [ABSTRACT FROM AUTHOR]

IntroductionPersistent Tic Disorders such as Tourette Syndrome are common neurodevelopmental disorders that are highly stigmatized. Many individuals with Persistent Tic Disorders experience peer rejection, loneliness, and self-stigma. Experiencing stigmatization during childhood can influence the persistence of moderate-to-severe tics later in life. Additionally, these factors have been associated with increased suicidal ideation, suicide attempts, and psychiatric symptom severity. There is a need for interventions to reduce stigma and stigmatization in Persistent Tic Disorders. Before developing cost-effective interventions to mitigate stigma’s profound downstream health impacts, a reliable measure of stigmatization must be created. The overarching goal of this research is to develop and validate the Tourette Discrimination-Stigmatization (TD-STIGMA) Scale.MethodsThis paper presents the study protocol for developing and validating the TD-STIGMA Scale. The study is designed as a mixed methods study to develop the TD-STIGMA scale and evaluate its psychometric properties. The study uses a phased approach: (1) collection of narrative and thematic content data through in-depth qualitative interviews of stakeholders, (2) development of a novel TD-STIGMA self-report scale using the Delphi Method based on these results, and (3) completion of analyses to determine the scale’s psychometric properties (confirmatory factor analysis, convergent, known-group, criterion validity, and test–retest reliability).DiscussionThis project will result in a personalized approach to stigma measurement about youth and young adults with Persistent Tic Disorders, which to date does not exist. There are several limitations. Comorbidities or spiritual or cultural beliefs may affect perceptions of stigma and are not directly assessed in this study. We will utilize institutional resources for community outreach to purposefully sample underrepresented minorities who may be at disproportionate risk of adverse outcomes. However, this may not be fully representative of the generalized tic population. The study team will be purposeful in maintaining participant engagement for study retention. Lastly, participants from a tertiary referral center may not fully represent the generalized tic community. However, we hope our broad recruitment strategy and virtual study visits will facilitate a diverse and inclusive sampling of the patient population.

Abstract Background Comprehensive Behavioral Intervention for Tics (CBIT) is a first-line treatment for tic disorders that aims to improve controllability over tics that an individual finds distressing or impairing. However, it is only effective for approximately half of patients. Supplementary motor area (SMA)-directed neurocircuitry plays a strong role in motor inhibition, and activity in this region is thought to contribute to tic expression. Targeted modulation of SMA using transcranial magnetic stimulation (TMS) may increase CBIT efficacy by improving patients' ability to implement tic controllability behaviors. Methods The CBIT + TMS trial is a two-phase, milestone-driven early-stage randomized controlled trial. The trial will test whether augmenting CBIT with inhibitory, non-invasive stimulation of SMA with TMS modifies activity in SMA-mediated circuits and enhances tic controllability in youth ages 12–21 years with chronic tics. Phase 1 will directly compare two rTMS augmentation strategies (1 Hz rTMS vs. cTBS) vs. sham in N = 60 participants. Quantifiable, a priori “Go/No Go Criteria” guide the decision to proceed to phase 2 and the selection of the optimal TMS regimen. Phase 2 will compare the optimal regimen vs. sham and test the link between neural target engagement and clinical outcomes in a new sample of N = 60 participants. Discussion This clinical trial is one of few to date testing TMS augmentation of therapy in a pediatric sample. The results will provide insight into whether TMS is a potentially viable strategy for enhancing CBIT efficacy and reveal potential neural and behavioral mechanisms of change. Trial registration ClinicalTrials.gov NCT04578912 . Registered on October 8, 2020.

This is the ninth yearly article in the Tourette Syndrome Research Highlights series, summarizing selected research reports from 2022 relevant to Tourette syndrome. The authors briefly summarize reports they consider most important or interesting.

Background: Comprehensive Behavioral Intervention for Tics (CBIT) is a first-line treatment for tic disorders that aims to improve controllability over tics that an individual finds distressing or impairing. However, it is only effective for approximately half of patients. Supplementary motor area (SMA)-directed neurocircuitry plays a strong role in motor inhibition, and activity in this region is thought to contribute to tic expression. Targeted modulation of SMA using transcranial magnetic stimulation (TMS) may increase CBIT efficacy by improving patients' ability to implement tic controllability behaviors. Methods: The CBIT + TMS trial is a two-phase, milestone-driven early-stage randomized controlled trial. The trial will test whether augmenting CBIT with inhibitory, non-invasive stimulation of SMA with TMS modifies activity in SMA-mediated circuits and enhances tic controllability in youth ages 12–21 years with chronic tics. Phase 1 will directly compare two rTMS augmentation strategies (1 Hz rTMS vs. cTBS) vs. sham in N = 60 participants. Quantifiable, a priori "Go/No Go Criteria" guide the decision to proceed to phase 2 and the selection of the optimal TMS regimen. Phase 2 will compare the optimal regimen vs. sham and test the link between neural target engagement and clinical outcomes in a new sample of N = 60 participants. Discussion: This clinical trial is one of few to date testing TMS augmentation of therapy in a pediatric sample. The results will provide insight into whether TMS is a potentially viable strategy for enhancing CBIT efficacy and reveal potential neural and behavioral mechanisms of change. Trial registration: ClinicalTrials.gov NCT04578912. Registered on October 8, 2020. [ABSTRACT FROM AUTHOR]

This is the ninth yearly article in the Tourette Syndrome Research Highlights series, summarizing selected research reports from 2022 relevant to Tourette syndrome. The authors briefly summarize reports they consider most important or interesting.

The article describes classification, aetiology and features of typical tic disorders (Tourette syndrome) and their management with an additional focus on a surge in atypical tic presentations following the COVID-19 pandemic, often described as functional tics, or functional tic-like movements. We discuss what explains their atypical nature and what might underpin this increase in incidence. Lastly the article provides an overview of management of functional tics, so readers can understand how management of these differs from typical tic disorders. [ABSTRACT FROM AUTHOR]

Objective: To provide an evidence-based review of the Comprehensive Behavioral Intervention for Tic (CBIT) disorders. Results: For close to a century, behavioral interventions for managing tics associated with Tourette and other tic disorders (TDs) were incorrectly considered ineffective and dangerous by the professional community, due, in large part, to unfounded fears that efforts to suppress tics would lead to a host of negative psychological, and even physical, outcomes (e.g., symptom substitution, tic rebound). Spurred by a growing body of research to the contrary, the Comprehensive Behavioral Treatment for Tics (CBIT) was developed to provide a tolerable and effective nonpharmacological treatment option, alone or in combination with medication, for youth and adults with tics associated with Tourette or other TDs. CBIT combines two evidence-based practices, habit reversal training (HRT) to address the urge-tic relationship and a functional intervention to identify and neutralize tic-related environmental factors. Based on positive findings from two large-scale randomized controlled trials that involved a total of 248 8-69-year olds with Tourette or chronic TD, CBIT has been designated as a first-line treatment, when available, for treating tics by the American Academy of Neurology and the European and Canadian medical academies. Conclusions: CBIT has demonstrated acute and durable efficacy when delivered alone or in combination with medication, in person, or via telehealth, and in the presence or absence of common comorbid conditions. Additional research is needed to develop and test treatment guidelines for the use of CBIT in combination with pharmacologic, neuromodulatory, and other intervention modalities.

Abstract Background It is unclear how to best measure the complex symptom presentation of pediatric acute-onset neuropsychiatric syndrome (PANS). Methods Well-characterized participants of a 2–5 year follow-up study (n = 34; 56% male) underwent clinical evaluations and completed scales assessing global symptom severity, functional impairment and specific psychiatric symptoms. We explored inter-correlations between the measures and used intraclass correlation coefficients to evaluate the agreement between clinician-, parent- and child ratings of the same constructs. Results Ratings on symptom-specific measures varied largely between participants. Agreement between informants was excellent on functional scales, fair-to-moderate on global severity scales and mixed on symptom-specific scales. Clinician-rated global and functional measures had stronger inter-correlations with parent- and child-rated functional measures than with symptom-specific measures. Conclusions General instruments assessing global severity and functioning are well suited for the assessment and follow-up of PANS, but should be complemented by symptom-specific scales representative of core symptoms.

This study investigated whether vitamin D is associated with the presence or severity of chronic tic disorders and their psychiatric comorbidities. This cross-sectional study compared serum 25-hydroxyvitamin D [25(OH)D] (ng/ml) levels among three groups: children and adolescents (3–16 years) with CTD (n = 327); first-degree relatives (3–10 years) of individuals with CTD who were assessed for a period of up to 7 years for possible onset of tics and developed tics within this period (n = 31); and first-degree relatives who did not develop tics and were ≥ 10 years old at their last assessment (n = 93). The relationship between 25(OH)D and the presence and severity of tics, as well as comorbid obsessive–compulsive disorder (OCD) and attention-deficit/hyperactivity disorder (ADHD), were analysed controlling for age, sex, season, centre, latitude, family relatedness, and comorbidities. When comparing the CTD cohort to the unaffected cohort, the observed result was contrary to the one expected: a 10 ng/ml increase in 25(OH)D was associated with higher odds of having CTD (OR 2.08, 95% CI 1.27–3.42, p < 0.01). There was no association between 25(OH)D and tic severity. However, a 10 ng/ml increase in 25(OH)D was associated with lower odds of having comorbid ADHD within the CTD cohort (OR 0.55, 95% CI 0.36–0.84, p = 0.01) and was inversely associated with ADHD symptom severity (β = − 2.52, 95% CI − 4.16–0.88, p < 0.01). In conclusion, lower vitamin D levels were not associated with a higher presence or severity of tics but were associated with the presence and severity of comorbid ADHD in children and adolescents with CTD. [ABSTRACT FROM AUTHOR]

Background: Gilles de la Tourette Syndrome (GTS) is a childhood-onset neuropsychiatric disorder characterised by motor and vocal tics. While Comprehensive Behavioural Intervention for Tics (CBIT) is an effective, non-pharmacological treatment for patients with GTS, the underlying neurophysiological basis of this intervention has not been investigated. Methods: To investigate the clinical effectiveness of CBIT in reducing tic severity in young people with GTS and explore neurophysiological mechanisms associated with clinical change. Results: There was a significant overall improvement in tic severity of large effect size. The Cortical Silent Period (CSP) to motor evoked potential (MEP) ratio (CSP/MEP ratio) increased after the intervention with a small effect size. Other neurophysiological measures of inhibition were not significantly related to the change in tic severity. Conclusions: Alongside significant clinical improvements, these results suggest a role for motor cortical Gamma-aminobutyric acid (GABA)-ergic inhibitory circuitry in the neurophysiological changes underlying CBIT treatment. These findings need to be replicated in larger studies using control samples. [ABSTRACT FROM AUTHOR]

Objective: To determine the long-term durability of behavior therapy for tics among youth with Tourette disorder and persistent (chronic) motor or vocal tic disorders.Method: Of the 126 youth who participated in a randomized controlled trial of behavior therapy 11 years prior, 80 were recruited for this longitudinal follow-up. Consenting participants were interviewed in person or remotely (Web-based video) by trained evaluators to determine the course of tics, current tic severity, and tic-related impairment. Recruitment and data collection occurred between 2014 and 2019, with an average follow-up duration of 11.2 years.Results: Treatment responders to both conditions in the original trial achieved partial, but not full, tic remission. Tic severity also decreased significantly across the sample, with 40% reporting partial remission. Behavior therapy responders (n = 21) in the original trial were more likely (67%) to achieve remission at follow-up (Total Tic Score = 12.52, SD = 10.75) compared to psychoeducation/supportive therapy responders (n = 6, 0%) at follow-up (Total Tic Score = 20.67, SD = 6.92) on the Yale Global Tic Severity Scale. Tic-related impairment decreased across the sample, with no significant differences between treatment groups or responders.Conclusion: Despite limitations of unmeasured variables and veracity of self-report at follow-up, this study supports guidelines recommending behavior therapy as the first-line intervention for tics. Further investigation of behavior therapy as an early preventive intervention also merits attention. [ABSTRACT FROM AUTHOR]

BackgroundThere has been a rise in explosive onset of tic-like behaviors during the COVID-19 pandemic. Historically, this is an uncommon phenomenology of functional movement disorders across all ages. Both the psychological burden of the pandemic and social media usage have been implicated in the rise of these tic-like behaviors.MethodsThis paper provides a narrative review of the literature on chronic tic disorders, functional tics, and mass functional illness with particular focus on the key distinguishing features, role of social media, and the role of COVID-19.ResultsThe COVID-19 pandemic has profoundly affected the mental health of many individuals, including children, adolescents, and their caregivers. Implementation of lockdowns, lifestyle disruptions, school closures, and social distancing have driven a surge in social media and digital technology use. The combination of predisposing factors, the psychological burden of the COVID-19 pandemic, and social media are implicated in the rise and spread of tic-like behaviors; which may represent a modern-day form of mass functional illness. While many of the features overlap with functional tics, there are emerging distinctive features that are important to recognize. A more encompassing term, Functional Tic-Like Behaviors, is used to better reflect multiple contributing factors.ConclusionKnowledge of these differences is essential to mitigate downstream health effects and poor outcomes.

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Background: There is clear evidence that tic disorders (TDs) are associated with psychosocial stress as well as emotional and behavioral problems. Studies have shown that individuals with TDs have higher acute physiological stress responses to external, single stressors (as reflected by saliva cortisol). The aim of the present study was to examine a physiological marker of longer-term stress (as reflected by hair cortisol concentration) in children and adolescents with TDs and unaffected siblings of individuals with TDs. Methods: Two samples of a European cohort were included in this study. In the COURSE sample, 412 children and adolescents aged 3–16 years with a chronic TD including Tourette syndrome according to DSM IV-TR criteria were included. The ONSET sample included 131 3–10 years old siblings of individuals with TDs, who themselves had no tics. Differences in hair cortisol concentration (HCC) between the two samples were examined. Within the COURSE sample, relations of HCC with tic severity and perceived psychosocial stress as well as potential effects and interaction effects of comorbid emotional and behavioral problems and psychotropic medication on HCC were investigated. Results: There were no differences in HCC between the two samples. In participants with TDs, there were no associations between HCC and tic severity or perceived psychosocial stress. No main effects of sex, psychotropic medication status and comorbid emotional and behavioral problems on HCC were found in participants with TDs. Conclusion: A link between HCC and TDs is not supported by the present results. [ABSTRACT FROM AUTHOR]

Little is known about the long-term prognosis of children with pediatric acute-onset neuropsychiatric syndrome (PANS). Out of the 46 eligible patients from the Karolinska PANS cohort, 34 consented to participate in a follow-up (median 3.3 years). Participants underwent a thorough clinical evaluation and were classified according to their clinical course. Resulting groups were compared on clinical characteristics and laboratory test results. We observed significant reductions in clinician-rated PANS symptom severity and improved general function. Two patients were classified as remitted, 20 as relapsing–remitting, and 12 as having a chronic-static/progressive course. The latter group had an earlier onset, greater impairment and received more pharmacological and psychological treatments. Although remission was rare, the majority of children with PANS were significantly improved over the follow-up period but a non-negligible minority of patients displayed a chronic-static/progressive course and required additional treatments. The proposed definitions of flare and clinical course may be useful in future clinical trials. [ABSTRACT FROM AUTHOR]

This document is a reply to a letter to the editor regarding the Rush Video-Based Tic Rating Scale-Revised (MRVS-R). The authors respond to critiques and suggestions made by Becker et al. They explain that they do not favor adding explicit instructions to patients not to suppress their tics, as it may draw attention to the tics. They also clarify that the instructions for determining blink rate do not suppress eye tics. The authors address other concerns raised by Becker et al and emphasize the importance of a practical and standardized protocol for assessing tics. [Extracted from the article]

The article discusses the Modified Rush Video-Based Tic Rating Scale (MRVS) and proposes changes to improve its standardization. However, the authors of the article raise concerns about some of the proposed changes, such as the lack of an instruction to not suppress tics and the reduction of rating time, which may result in the underrepresentation of tics. They also question the validity of the comparison between tic counts from different intervals and the increase in internal consistency for the revised score. Additionally, the authors argue that a high convergent validity between the MRVS and the Yale Global Tic Severity Scale (YGTSS) is not desirable, as they assess different aspects of the condition. Overall, the authors recommend further evaluation and reconsideration of the suggested changes. [Extracted from the article]

Background: It is unclear how to best measure the complex symptom presentation of pediatric acute-onset neuropsychiatric syndrome (PANS). Methods: Well-characterized participants of a 2–5 year follow-up study (n = 34; 56% male) underwent clinical evaluations and completed scales assessing global symptom severity, functional impairment and specific psychiatric symptoms. We explored inter-correlations between the measures and used intraclass correlation coefficients to evaluate the agreement between clinician-, parent- and child ratings of the same constructs. Results: Ratings on symptom-specific measures varied largely between participants. Agreement between informants was excellent on functional scales, fair-to-moderate on global severity scales and mixed on symptom-specific scales. Clinician-rated global and functional measures had stronger inter-correlations with parent- and child-rated functional measures than with symptom-specific measures. Conclusions: General instruments assessing global severity and functioning are well suited for the assessment and follow-up of PANS, but should be complemented by symptom-specific scales representative of core symptoms. [ABSTRACT FROM AUTHOR]

Tourette syndrome (TS) is a severe neuropsychiatric disorder characterized by recurrent, involuntary physical and verbal tics. With a prevalence as high as 1% in children, a deeper understanding of the etiology of the disorder and contributions to risk is critical. Here, we cover the current body of knowledge in scientific literature regarding the genetics of TS. We first review the history and diagnostic criteria for TS cases. We then cover the prevalence, and begin to address the etiology of the disorder. We highlight long-standing evidence for a genetic contribution to TS risk from epidemiology studies focused on twins, families, and population-scale data. Finally, we summarize current large-scale genetic studies of TS along specific classes of genetic variation, including common variation, rare copy number variation, and de novo variation that impact protein-coding sequence. Although these variants do not account for the entirety of TS genetic risk, current evidence is clear that each class of variation is a factor in the overall risk architecture across TS cases. [ABSTRACT FROM AUTHOR]

Background: Persistent motor or vocal tic disorder (PMVT) has been hypothesized to be a forme fruste of Tourette syndrome (TS). Although the primary diagnostic criterion for PMVT (presence of motor or vocal tics, but not both) is clear, less is known about its clinical presentation. Objective: The goals of this study were to compare the prevalence and number of comorbid psychiatric disorders, tic severity, age at tic onset, and family history for TS and PMVT. Methods: We analyzed data from two independent cohorts using generalized linear equations and confirmed our findings using meta‐analyses, incorporating data from previously published literature. Results: Rates of obsessive–compulsive disorder (OCD) and attention deficit hyperactivity disorder (ADHD) were lower in PMVT than in TS in all analyses. Other psychiatric comorbidities occurred with similar frequencies in PMVT and TS in both cohorts, although meta‐analyses suggested lower rates of most psychiatric disorders in PMVT compared with TS. ADHD and OCD increased the odds of comorbid mood, anxiety, substance use, and disruptive behaviors, and accounted for observed differences between PMVT and TS. Age of tic onset was approximately 2 years later, and tic severity was lower in PMVT than in TS. First‐degree relatives had elevated rates of TS, PMVT, OCD, and ADHD compared with population prevalences, with rates of TS equal to or greater than PMVT rates. Conclusions: Our findings support the hypothesis that PMVT and TS occur along a clinical spectrum in which TS is a more severe and PMVT a less severe manifestation of a continuous neurodevelopmental tic spectrum disorder. © 2021 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society [ABSTRACT FROM AUTHOR]

Objective: Children needing admission to an inpatient mental health unit often present with severe neuropsychiatric disorders characterised by complex psychopathology. We aimed to examine all admitted children with comorbid chronic tic disorder (CTD) and Tourette syndrome (TS) over a 10-year period and determine the clinical significance of these diagnoses. Method: A retrospective, naturalistic study was conducted, comparing children with and without CTD/TS in terms of co-morbid diagnoses, medication use, access to education, aggression contributing to the admission, duration of admission, functional outcomes and satisfaction with treatment. Data were analysed using Chi-square/Fisher's exact test and t -test for categorical and continuous variables, respectively, and subsequently with unadjusted and adjusted linear and logistic regression analyses. Results: A relatively high proportion of children had co-morbid CTD/TS (19.7%). There was a significant association with co-morbid obsessive-compulsive disorder, intellectual disability and autism spectrum disorder but not attention deficit hyperactivity disorder. CTD/TS were associated with longer admissions even after adjustments for confounding but did not seem to be independently associated with other examined clinical characteristics. Conclusions: The prevalence of CTD/TS in children needing inpatient treatment is significant. In our sample, comorbid CTD/TS seem to represent a marker of overall symptom severity as evidenced by longer admissions. [ABSTRACT FROM AUTHOR]

We compared anxiety symptoms in youth with and without tic disorders by comparing scores on the Multidimensional Anxiety Scale for Children (MASC) in youth with tic disorders to those in a concurrent community control group and in a group of treatment-seeking anxious youth from the Child/Adolescent Anxiety Multimodal Study (CAMS). Data from 176 youth with tic disorders, 93 control subjects, and 488 CAMS participants were included. Compared to youth with tic disorders, controls had lower total MASC scores (p < 0.0001) and CAMS participants had similar total MASC scores (p = 0.13). Separation Anxiety (p = 0.0003) and Physical Symptom (p < 0.0001) subscale scores were higher in youth with tic disorders than in CAMS participants. We conclude that the anxiety symptom profile differs in youth with and without tic disorders, which may have important implications for targeting treatment of anxiety in youth with tic disorders. [ABSTRACT FROM AUTHOR]

Functional impairment is an important factor in Tic Disorder treatment decisions. We evaluated the mini Child Tourette Syndrome Impairment Scale (mini-CTIM) for correlation with symptom severity and association with interventions. A total of 61 randomly selected tic encounters were retrospectively analyzed for mini-CTIM correlation with symptom severity scores and compared between patients who received treatment and those who did not. Regression models identified factors associated with treatment decisions. Mini-CTIM-tic scores correlated with tic severity and mini-CTIM-non-tic scores correlated with attention-deficit hyperactivity disorder (ADHD) severity. Tic treatment was associated with higher child, but not parent, mini-CTIM-tic scores. Regression models identified that comorbidity treatment was predicted by ADHD severity, obsessive compulsive disorder severity, and parent but not child mini-CTIM-non-tic scores. These findings suggest children have valuable insight into their tic-related impairment, but parent assessment is important for evaluating comorbidity-related impairment. The mini-CTIM may be a useful clinical tool for assessing tic-related impairment. [ABSTRACT FROM AUTHOR]

Tics are unique from most movement disorders, in that they are partially suppressible. As part of the inhibitory motor network, the pre-supplementary motor area is engaged in motor control and may be involved in tic physiology. We used dual-site transcranial magnetic stimulation to assess inhibitory connectivity between right pre-supplementary motor area and left primary motor cortex, which has previously been demonstrated in healthy adults. We also used diffusion tensor imaging to investigate white matter connectivity in children with chronic tics. Twelve children with chronic tic disorder and fourteen typically developing controls underwent MRI with diffusion tensor imaging indices analysis followed by single and paired-pulse transcranial magnetic stimulation with conditioning pulse over the right pre-supplementary motor area followed by left motor cortex test pulse. Neurophysiologic and imaging data relationships to measures of tic severity and suppressibility were also evaluated in tic patients. Pre-supplementary motor area-mediated inhibition of left motor cortex was present in healthy control children but not in chronic tic disorder participants. Less inhibition correlated with worse tic suppressibility (ρ = − 0.73, p = 0.047). Imaging analysis showed increased fractional anisotropy in the right superior longitudinal fasciculus, corpus callosum, corona radiata and posterior limb of the internal capsule (p < 0.05) in tic participants, which correlated with lower self-reported tic suppressibility (ρ = − 0.70, p = 0.05). Physiologic data revealed impaired frontal-mediated motor cortex inhibition in chronic tic participants, and imaging analysis showed abnormalities in motor pathways. Collectively, the neurophysiologic and neuroanatomic data correlate with tic suppressibility, supporting the relevancy to tic pathophysiology. [ABSTRACT FROM AUTHOR]

Neuropsychiatric disorders are highly prevalent conditions with significant individual, societal, and economic impacts. A major challenge in the diagnosis and treatment of these conditions is the lack of sensitive, reliable, objective, quantitative tools to inform diagnosis, and measure symptom severity. Currently available assays rely on self-reports and clinician observations, leading to subjective analysis. As a step toward creating quantitative assays of neuropsychiatric symptoms, we propose an immersive environment to track behaviors relevant to neuropsychiatric symptomatology and to systematically study the effect of environmental contexts on certain behaviors. Moreover, the overarching theme leads to connected tele-psychiatry which can provide effective assessment.

Gilles de la Tourette syndrome (GTS) can manifest as debilitating, medically-refractory tics for which deep brain stimulation (DBS) of the centromedian-parafascicular complex (CM) can provide effective treatment. However, patients have reported benefit with activation of contacts dorsal to the CM and likely in the ventro-lateral thalamus (VL). At our institution, a case of a robust and durable response in a GTS patient required stimulation in the CM and more dorsally. We explore the structural connectivity of thalamic subregions associated with GTS using diffusion MRI tractography. Diffusion weighted images from 40 healthy Human Connectome Project (HCP) subjects and our GTS patient were analyzed. The VL posterior nucleus (VLp) and the CM were used as seeds for whole-brain probabilistic tractography. Leads were localized via linear registration of pre-/post-operative imaging and cross-referenced with the DBS Intrinsic Template Atlas. Tractography revealed high streamline probability from the CM and VLp to the superior frontal gyrus, rostral middle frontal gyrus, brainstem, and ventral diencephalon. Given reported variable responses to DBS along the thalamus, we segmented the VLp based on its connectivity profile. Ventral and dorsal subdivisions emerged, with streamline probability patterns differing between the dorsal VLp and CM. The CM, the most reported DBS target for GTS, and the dorsal VLp have different but seemingly complimentary connectivity profiles as evidenced by our patient who, at 1-year post-operatively, had significant therapeutic benefit. Stimulation of both regions may better target reward and motor circuits, resulting in enhanced symptom control for GTS. [ABSTRACT FROM AUTHOR]

Background: Tourette syndrome (TS) manifests with motor and vocal tics that can reach disabling intensity. Established therapies may show insufficient relief or side effects. Cannabinoids have demonstrated therapeutic potential in small studies. This report presents buccal Nabiximols "as required" in the treatment of tics in TS. Case report: A 25-year-old man presented with stigmatizing motor and phonic tics after cessation of daily Cannabis use. After Tiaprid 300 mg per day had shown no sufficient effect a trial of Nabiximols reduced tics by >90%. Discussion: Nabiximols could be an adjunct treatment in TS for situations were tics are severly disabling. [ABSTRACT FROM AUTHOR]

Objective: The goal of this article is to review the past decade's literature and provide a critical commentary on the involvement of immunological mechanisms in normal brain development, as well as its role in the pathophysiology of Tourette syndrome, other Chronic tic disorders (CTD), and related neuropsychiatric disorders including Obsessive-compulsive disorder (OCD) and Attention deficit hyperactivity disorder (ADHD).Methods: We conducted a literature search using the Medline/PubMed and EMBASE electronic databases to locate relevant articles and abstracts published between 2009 and 2020, using a comprehensive list of search terms related to immune mechanisms and the diseases of interest, including both clinical and animal model studies.Results: The cellular and molecular processes that constitute our “immune system” are crucial to normal brain development and the formation and maintenance of neural circuits. It is also increasingly evident that innate and adaptive systemic immune pathways, as well as neuroinflammatory mechanisms, play an important role in the pathobiology of at least a subset of individuals with Tourette syndrome and related neuropsychiatric disorders In the conceptual framework of the holobiont theory, emerging evidence points also to the importance of the “microbiota-gut-brain axis” in the pathobiology of these neurodevelopmental disorders.Conclusions: Neural development is an enormously complex and dynamic process. Immunological pathways are implicated in several early neurodevelopmental processes including the formation and refinement of neural circuits. Hyper-reactivity of systemic immune pathways and neuroinflammation may contribute to the natural fluctuations of the core behavioral features of CTD, OCD, and ADHD. There is still limited knowledge of the efficacy of direct and indirect (i.e., through environmental modifications) immune-modulatory interventions in the treatment of these disorders. Future research also needs to focus on the key molecular pathways through which dysbiosis of different tissue microbiota influence neuroimmune interactions in these disorders, and how microbiota modification could modify their natural history. It is also possible that valid biomarkers will emerge that will guide a more personalized approach to the treatment of these disorders.

Tourette ‘s syndrome (TS) currently has very limited FDA approved treatment options, despite the fact that TS and other pediatric tic disorders are not uncommon and can cause significant emotional and physical distress for patients and their families. For providers who regularly see these conditions in practice this inadequacy is highly frustrating on the treatment side as there are such limited options to offer to these families, and the outcome to hope for is not highly optimistic. The outcome of this case presentation is an example of how health care providers can use clinical knowledge in combination with evidence-based practice to advocate for their patients and find new solutions that could be, and in this case were, life changing. It promotes providers to advocate for their patients and expand their knowledge. The final medication treatment utilized in this case is currently off label, however the method of finding and obtaining the treatment explained in the case additionally reveal to readers how to cautiously but judiciously make off label treatment decisions that can be important to practice and patient outcomes. In the course of this case the off-label medication use decision was backed by scientific literature and ongoing FDA studies as well as consulting with a collaborating psychiatrist. The subject matter and method of reasoning within this case also promote psychiatric providers use of evidence-based practice and implementation of science into active practice.

Objective: The goal of this article is to review the past decade's literature and provide a critical commentary on the involvement of immunological mechanisms in normal brain development, as well as its role in the pathophysiology of Tourette syndrome, other Chronic tic disorders (CTD), and related neuropsychiatric disorders including Obsessive-compulsive disorder (OCD) and Attention deficit hyperactivity disorder (ADHD). Methods: We conducted a literature search using the Medline/PubMed and EMBASE electronic databases to locate relevant articles and abstracts published between 2009 and 2020, using a comprehensive list of search terms related to immune mechanisms and the diseases of interest, including both clinical and animal model studies. Results: The cellular and molecular processes that constitute our "immune system" are crucial to normal brain development and the formation and maintenance of neural circuits. It is also increasingly evident that innate and adaptive systemic immune pathways, as well as neuroinflammatory mechanisms, play an important role in the pathobiology of at least a subset of individuals with Tourette syndrome and related neuropsychiatric disorders In the conceptual framework of the holobiont theory, emerging evidence points also to the importance of the "microbiota-gut-brain axis" in the pathobiology of these neurodevelopmental disorders. Conclusions: Neural development is an enormously complex and dynamic process. Immunological pathways are implicated in several early neurodevelopmental processes including the formation and refinement of neural circuits. Hyper-reactivity of systemic immune pathways and neuroinflammation may contribute to the natural fluctuations of the core behavioral features of CTD, OCD, and ADHD. There is still limited knowledge of the efficacy of direct and indirect (i.e., through environmental modifications) immune-modulatory interventions in the treatment of these disorders. Future research also needs to focus on the key molecular pathways through which dysbiosis of different tissue microbiota influence neuroimmune interactions in these disorders, and how microbiota modification could modify their natural history. It is also possible that valid biomarkers will emerge that will guide a more personalized approach to the treatment of these disorders. [ABSTRACT FROM AUTHOR]

Children with Tourette syndrome often have behavioral and social difficulties, which may be associated with co-occurring mental, emotional, or behavioral disorders. This study investigated social competence, including behavioral problems and social skills, and social activities between children with and without Tourette syndrome using a nationally representative sample. In the 2007 National Survey of Children's Health, parents reported on health care provider diagnosis of Tourette syndrome, co-occurring mental, emotional, or behavioral disorders, and indicators of social competence. Children aged 6-17 years with and without Tourette syndrome were compared. Most (78.7%) children with Tourette syndrome had a co-occurring mental, emotional, or behavioral disorder. Children with Tourette syndrome had significantly lower social competence, exhibited by higher levels of behavior problems (mean score 11.6 for Tourette syndrome and 9.0 for no Tourette syndrome) and lower levels of social skills (mean 15.3) than children without a Tourette syndrome diagnosis (mean 17.1); however, these associations were no longer significant after controlling for co-occurring mental, emotional, or behavioral disorders. Moderate to severe Tourette syndrome was associated with the highest ratings of behavioral problems and the lowest ratings of social skills. Children with and without Tourette syndrome were equally likely to participate in social activities; the difference for children with moderate to severe Tourette syndrome being less likely to participate in activities compared to children with mild Tourette syndrome had a chi-square test P value of.05. In conclusion, Tourette syndrome was associated with lower social competence, particularly for children with moderate to severe Tourette syndrome. Monitoring social functioning and co-occurring conditions among children with Tourette syndrome, and referral for evidence-based interventions when needed, may benefit overall health and functioning. [ABSTRACT FROM AUTHOR]

Tourette Syndrome (TS) is a childhood-onset neurobehavioral disorder, which is characterized by motor and sound tics. Current studies have identified some specific factors that lead to tic reduction, such as relaxation, concentration, musical activities, and the execution of voluntary and finalistic movements. Patients with TS show a higher level of creativity than other people, which can be channeled, together with their excessive motor energy, into various functional activities that favor the reduction of tics. Moreover, in the last decades, music has been used as a rehabilitative tool, since it has shown to induce a positive effect on TS patients' mood and to facilitate the performance of fluid and rhythmic voluntary movements. The present study refers to "Imagine, Tourette!", a motor imagery, music-based intervention aimed at reducing the manifestation of motor and sound tics in adult TS patients. To test the specific effect of such a kind of training, 8 TS patients were exposed to one of two interventions: the experimental group performed motor tasks based on music whereas the control group performed motor tasks without the accompaniment of music. The hypothesis tested was that performing motor tasks accompanied by music could reduce the severity of tics and have an effect on patients' mood more than simply performing motor tasks alone. The results supported the hypothesis: music played a specific effect on the manifestation of tics and on the patients' mood, confirming its potentially positive role in motor interventions addressed to TS. [ABSTRACT FROM AUTHOR]

Obsessive-compulsive disorder (OCD) is a debilitating neuropsychiatric disorder with a genetic risk component, yet identification of high-confidence risk genes has been challenging. In recent years, risk gene discovery in other complex psychiatric disorders has been achieved by studying rare de novo (DN) coding variants. We performed whole-exome sequencing in 222 OCD parent-child trios (184 trios after quality control), comparing DN variant frequencies with 777 previously sequenced unaffected trios. We estimated the contribution of DN mutations to OCD risk and the number of genes involved. Finally, we looked for gene enrichment in other datasets and canonical pathways. DN likely gene disrupting and predicted damaging missense variants are enriched in OCD probands (rate ratio, 1.52; p =.0005) and contribute to risk. We identified 2 high-confidence risk genes, each containing 2 DN damaging variants in unrelated probands: CHD8 and SCUBE1. We estimate that 34% of DN damaging variants in OCD contribute to risk and that DN damaging variants in approximately 335 genes contribute to risk in 22% of OCD cases. Furthermore, genes harboring DN damaging variants in OCD are enriched for those reported in neurodevelopmental disorders, particularly Tourette's disorder and autism spectrum disorder. An exploratory network analysis reveals significant functional connectivity and enrichment in canonical pathways, biological processes, and disease networks. Our findings show a pathway toward systematic gene discovery in OCD via identification of DN damaging variants. Sequencing larger cohorts of OCD parent-child trios will reveal more OCD risk genes and will provide needed insights into underlying disease biology. [ABSTRACT FROM AUTHOR]

Objective: To examine parent's perceptions of deep brain stimulation (DBS) and whether DBS is perceived to be a viable and safe treatment for their adolescent child presenting with a severe, treatment-resistant neurological or psychiatric condition. Method: Two hundred and seventy-nine parents completed an online survey using Amazon Mechanical Turk (MTurk). Participants were presented with five vignette scenarios involving adolescents with severe, treatment-resistant neurological or psychiatric conditions: Rett syndrome, autism spectrum disorder, epilepsy, obsessive-compulsive disorder, and Tourette syndrome. Parents were then asked to evaluate each scenario and rate overall acceptability of using DBS to improve their child's core symptoms. Data were collected over a period of 2 weeks in the month of October 2018. Results: We found that parents reported favorable impressions of DBS regardless of the target condition, especially when greater improvement could be assured and when their child had the capacity to assist in the treatment decision-making. Parents indicated some reluctance to use DBS when possible safety concerns were present. Familiarity with DBS was directly associated with attitudes. Conclusions: The findings highlight an overall parental willingness to consider DBS as a treatment option for key symptoms of neurological and psychiatric conditions in adolescents. [ABSTRACT FROM AUTHOR]

The defining character of tics is that they can be transiently suppressed by volitional effort of will, and at a behavioural level this has led to the concept that tics result from a failure of inhibition. However, this logic conflates the mechanism responsible for the production of tics with that used in suppressing them. Volitional inhibition of motor output could be increased to prevent the tic from reaching the threshold for expression, although this has been extensively investigated with conflicting results. Alternatively, automatic inhibition could prevent the initial excitation of the striatal tic focus-a hypothesis we have previously introduced. To reconcile these competing hypotheses, we examined different types of motor inhibition in a group of 19 patients with primary tic disorders and 15 healthy volunteers. We probed proactive and reactive inhibition using the conditional stop-signal task, and applied transcranial magnetic stimulation to the motor cortex, to assess movement preparation and execution. We assessed automatic motor inhibition with the masked priming task. We found that volitional movement preparation, execution and inhibition (proactive and reactive) were not impaired in tic disorders. We speculate that these mechanisms are recruited during volitional tic suppression, and that they prevent expression of the tic by inhibiting the nascent excitation released by the tic generator. In contrast, automatic inhibition was abnormal/impaired in patients with tic disorders. In the masked priming task, positive and negative compatibility effects were found for healthy controls, whereas patients with tics exhibited strong positive compatibility effects, but no negative compatibility effect indicative of impaired automatic inhibition. Patients also made more errors on the masked priming task than healthy control subjects and the types of errors were consistent with impaired automatic inhibition. Errors associated with impaired automatic inhibition were positively correlated with tic severity. We conclude that voluntary movement preparation/generation and volitional inhibition are normal in tic disorders, whereas automatic inhibition is impaired-a deficit that correlated with tic severity and thus may constitute a potential mechanism by which tics are generated. [ABSTRACT FROM AUTHOR]

Abstract Gilles de la Tourette syndrome (TS) is a complex developmental neuropsychiatric condition in which motor manifestations are often accompanied by comorbid conditions that impact the patient’s quality of life. In the DSM-5, TS belongs to the “neurodevelopmental disorders” group, together with other neurodevelopmental conditions, frequently co-occurring. In this study, we searched the PubMed database using a combination of keywords associating TS and all neurodevelopmental diagnoses. From 1009 original reports, we identified 36 studies addressing TS and neurodevelopmental comorbidities. The available evidence suggests the following: (1) neurodevelopmental comorbidities in TS are the rule, rather than the exception; (2) attention deficit/hyperactivity disorder (ADHD) is the most frequent; (3) there is a continuum from a simple (TS + ADHD or/and learning disorder) to a more complex phenotype (TS + autism spectrum disorder). We conclude that a prompt diagnosis and a detailed description of TS comorbidities are necessary not only to understand the aetiological basis of neurodevelopmental disorders but also to address specific rehabilitative and therapeutic approaches.