scispace - formally typeset
Search or ask a question

Showing papers on "Tourette syndrome published in 2022"


Journal ArticleDOI
TL;DR: The prevalence and clinical characteristics of children with rapid onset functional tic‐like behaviours during the COVID‐19 pandemic are reported.
Abstract: To report the prevalence and clinical characteristics of children with rapid onset functional tic‐like behaviours during the COVID‐19 pandemic.

28 citations


Journal ArticleDOI
TL;DR: In a longitudinal follow-up study, this paper found that behavior therapy was the first-line intervention for tics among youth with Tourette disorder and persistent (chronic) motor or vocal tic disorders.
Abstract: To determine the long-term durability of behavior therapy for tics among youth with Tourette disorder and persistent (chronic) motor or vocal tic disorders.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.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.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.

15 citations


Journal ArticleDOI
TL;DR: This prospective cohort study describes the course and treatment of rapid onset functional tic‐like behaviors in adolescents and adults previously reported in two case series.
Abstract: The prognosis of rapid onset functional tic‐like behaviors (FTLBs) is unknown. This prospective cohort study describes the course and treatment of rapid onset FTLBs in adolescents (n = 20) and adults (n = 9) previously reported in two case series.

13 citations


Journal ArticleDOI
TL;DR: In this article , the authors compared the clinical features between males and females with Tourette syndrome and chronic tic disorder, and found that males had a statistically higher frequency of complex motor tics and earlier age at onset than females in the multivariate regression analysis.
Abstract: Tourette syndrome (TS) is a neurodevelopmental disorder characterized by the presence of motor and phonic tics. It is at least three times more common in males compared with females; however, the clinical phenomenology between sexes has not been fully examined. We aimed to contrast the clinical features between males and females with TS and chronic tic disorder.We studied 201 consecutive patients fulfilling the diagnostic criteria for TS, persistent (or chronic) motor and vocal tic disorder and provisional tic disorder that were considered within the TS spectrum disorder. We performed blinded evaluations of video-recordings and retrospectively reviewed the clinical charts of all patients.Age ranges between 4 and 65 years. Males represented 77.6% of patients in the cohort. Overall, no differences were observed in the frequency, distribution and complexity of tics between sexes, except for a higher frequency of attention-deficit/hyperactivity disorder (ADHD) (P = .003) among males. Patients younger than 18-years old, in addition to a higher frequency of ADHD (P = .026), males had a statistically higher frequency of complex motor tics (P = .049) and earlier age at onset (P = .072) than females in the multivariate regression analysis. However, these differences were lost in patients older than 18 years, due to increased complexity of tics in females with aging.A sexual dimorphism was observed between patients with TS mainly before age of 18 years, suggesting an earlier onset of some types of tics and ADHD in males compared to females.

13 citations


Journal ArticleDOI
TL;DR: In this paper , an internet-delivered, completely therapist-independent CBIT intervention (iCBIT Minddistrict®) was compared to placebo and face-to-face (f2f) CBIT.
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 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.

11 citations


Journal ArticleDOI
TL;DR: In this article , the authors search the electronic database of PubMed for relevant studies using the search terms: ('Tourette syndrome' [MeSH Terms] OR 'Gilles de la Tourette syndrome', 'tic disorders' [MESH Terms], 'tic disorder', 'tics' [ MESH Terms' and 'tics disorder' (MeSH terms) and 'tic disease' [SH Terms) for the treatment of patients with Tourette Syndrome.
Abstract: Gilles de la Tourette syndrome (GTS) is a childhood onset disorder characterised by the presence of motor and vocal tics. The guidelines of both the American Academy of Neurology (AAN) as well as the European Society for the Study of Tourette Syndrome (ESSTS) recommend behavioural therapy and pharmacotherapy, mainly with antipsychotics, as first line treatments for tics. In spite of these well-established therapeutic approaches, a significant number of patients are dissatisfied because of insufficient tic reduction or intolerable side effects. Previous studies have suggested that cannabis-based medicine (CBM) might be an alternative treatment in these patients.Two reviewers (KS, NS) searched the electronic database of PubMed on 1 July, 2021 for relevant studies using the search terms: ('Tourette syndrome' [MeSH Terms] OR 'Gilles de la Tourette syndrome' [MeSH Terms] OR 'tic disorders' [MeSH Terms] OR 'tics' [MeSH Terms] OR 'tic disorders'[Title/Abstract]) AND ('cannabis-based medicine' [Title/Abstract] OR 'cannabis' [Title/Abstract] OR 'dronabinol' [Title/Abstract] OR 'nabiximols' [Title/Abstract] OR 'tetrahydrocannabinol' [Title/Abstract] OR 'THC' [Title/Abstract] OR 'cannabidiol' [Title/Abstract], limit: 'humans'. These studies were further reviewed for additional relevant citations. The titles and abstracts of the studies obtained through this search were examined by two reviewers (KS, NS) in order to determine article inclusion. Discrepancies were addressed by the reviewers through discussion and eventually conversation with the senior reviewer (KMV).Although the amount of evidence supporting the use of CBM in GTS is growing, the majority of studies are still limited to case reports, case series, and open uncontrolled studies. To date, only two small randomised controlled trials (RCTs) using tetrahydrocannabinol (THC, dronabinol) have been published demonstrating the safety and efficacy of this intervention in the treatment of tics in patients with GTS. On the other hand, another RCT with Lu AG06466 (formerly known as ABX-1431), a modulator of endocannabinoid neurotransmission, has failed to prove effective in the therapy of GTS. Accordingly, under the guidelines of both the ESSTS and the AAN, treatment with CBM is categorised as an experimental intervention that should be applied to patients who are otherwise treatment-resistant.Increasing evidence suggests that CBM is efficacious in the treatment of tics and psychiatric comorbidities in patients with GTS. The results of ongoing larger RCTs, such as CANNA-TICS (ClinicalTrials.gov Identifier: NCT03087201), will further clarify the role of CBM in the treatment of patients with GTS.

11 citations


Journal ArticleDOI
TL;DR: In this paper , a longitudinal study aimed to find clinical precursors of Tourette syndrome (TS) by assessing a range of clinical characteristics prior to tic onset in comparison with children without onset of tics.
Abstract: Children with Tourette syndrome (TS) often have comorbid disorders, particularly attention-deficit/hyperactivity disorder (ADHD) and obsessive-compulsive disorder (OCD). While subtle premorbid symptoms have been described in various psychiatric disorders, the presence of clinical precursors that may exist before the onset of tics is unknown. This longitudinal study aimed to find clinical precursors of tics by assessing a range of clinical characteristics prior to tic onset in comparison with children without onset of tics.A sample of 187 3- to 10-year-old first-degree unaffected relatives of children with TS were followed up to 7 years in the European Multicentre Tics in Children Study (EMTICS). We investigated whether clinical characteristics assessed at baseline predicted tic onset, comparing 126 children without tic onset to 61 children who developed tics. We used the least absolute shrinkage and selection operator (LASSO) method, a penalised logistic regression approach. We also explored sex differences and repeated our analyses in an age- and sex-matched subsample.Children with tic onset were more frequently male (β = -0.36), had higher baseline severity of conduct problems (β = 0.23), autism spectrum disorder symptoms (ASD; β = 0.08), compulsions (β = 0.02) and emotional problems (β = 0.03) compared to children without tic onset. Conduct and ASD problems were male-specific predictors, whereas severity of compulsions and oppositional (β = 0.39) and emotional problems were female-specific predictors.This study supports the presence of clinical precursors prior to tic onset and highlights the need of sex-specific monitoring of children at risk of developing tics. This may aid in the earlier detection of tics, particularly in females. We moreover found that tics most often persisted one year after tic onset, in contrast to the common belief that tics are mostly transient.

11 citations


Journal ArticleDOI
TL;DR: Support is found for the notion that premonitory sensory phenomena, sensory stimulation, and other environmental stressors that impact the HPA axis can influence tics through dopaminergic neurotransmission, and it is concluded that there is an emotional component to ticking behaviors.
Abstract: Tics can be associated with neurological disorders and are thought to be the result of dysfunctional basal ganglia pathways. In Tourette Syndrome (TS), excess dopamine in the striatum is thought to excite the thalamo-cortical circuits, producing tics. When external stressors activate the hypothalamic-pituitary-adrenal (HPA) axis, more dopamine is produced, furthering the excitation of tic-producing pathways. Emotional processing structures in the limbic are also activated during tics, providing further evidence of a possible emotional component in motor ticking behaviors. The purpose of this review is to better understand the relationship between emotional states and ticking behavior. We found support for the notion that premonitory sensory phenomena (PSP), sensory stimulation, and other environmental stressors that impact the HPA axis can influence tics through dopaminergic neurotransmission. Dopamine plays a vital role in cognition and motor control and is an important neurotransmitter in the pathophysiology of other disorders such as obsessive–compulsive disorder (OCD) and attention deficit hyperactivity disorder (ADHD), which tend to be comorbid with ticking disorders and are thought to use similar pathways. It is concluded that there is an emotional component to ticking behaviors. Emotions primarily involving anxiety, tension, stress, and frustration have been associated with exacerbated tics, with PSP contributing to these feelings.

10 citations


Journal ArticleDOI
TL;DR: The Mini-Child Tourette Syndrome Impairment Scale (mini-CTIM) is a validated clinical tool for assessing, in children and adolescents with tic disorders, tic- and non-tic-related impairment in home, school, and social settings as discussed by the authors .

8 citations


Journal ArticleDOI
TL;DR: Based on available prevalence estimates applied to 2020 population data from the U.S. Census, this article estimated that 350,000-450,000 children and adults have Tourette syndrome and about one million have other persistent tic disorders.
Abstract: Estimates of the number of people in the U.S. with Tourette syndrome or other persistent tic disorders can inform service provision planning. Based on available prevalence estimates applied to 2020 population data from the U.S. Census, we estimated that 350,000-450,000 U.S. children and adults have Tourette syndrome and about one million have other persistent tic disorders. Variation across studies makes estimating the total number of people in the United States affected by these disorders challenging. More precise measurement could ensure that prevalence estimates accurately reflect all who are impacted by these disorders and who could benefit from evidence-based services.

8 citations


Journal ArticleDOI
25 Feb 2022-Brain
TL;DR: In this paper , a group of Tourette syndrome researchers, clinicians, and individuals with tics detail concerns with Müller-Vahl et al.'s postulate that this phenomenon is a mass sociogenic illness.
Abstract: Recent reports from Tourette syndrome clinical researchers in North America and Europe1,2 describe a recent increase in young patients presenting to Tourette syndrome clinics. Reported commonalities in clinical presentation include a female preponderance, older age of first detected symptoms, complex behaviours (e.g. phrases, coprolalia, long/sequenced movements), significant functional impairment, and similarities to behaviours recorded in videos on social media platforms, notably TikTok. This has raised important questions about aetiology and how to best diagnose and treat these individuals. In their recent Brain paper, Müller-Vahl et al.3 postulated that this phenomenon is a ‘mass sociogenic illness.’ The function of this assertion could be to caution clinicians and patients against using interventions contraindicated for those with functional movement disorder (FMD). However, this postulate does not follow neatly from the current state of the evidence, and the rhetorical language used risks negatively impacting patients by implying that these symptoms are ‘attention seeking’ behaviours. In this response, written by a group of Tourette syndrome researchers, clinicians, and individuals with tics, we detail concerns with the paper.

Journal ArticleDOI
25 Feb 2022-Brain
TL;DR: In this article , a group of Tourette syndrome researchers, clinicians, and individuals with tics detail concerns with Müller-Vahl et al.'s postulate that this phenomenon is a mass sociogenic illness.
Abstract: Recent reports from Tourette syndrome clinical researchers in North America and Europe1,2 describe a recent increase in young patients presenting to Tourette syndrome clinics. Reported commonalities in clinical presentation include a female preponderance, older age of first detected symptoms, complex behaviours (e.g. phrases, coprolalia, long/sequenced movements), significant functional impairment, and similarities to behaviours recorded in videos on social media platforms, notably TikTok. This has raised important questions about aetiology and how to best diagnose and treat these individuals. In their recent Brain paper, Müller-Vahl et al.3 postulated that this phenomenon is a ‘mass sociogenic illness.’ The function of this assertion could be to caution clinicians and patients against using interventions contraindicated for those with functional movement disorder (FMD). However, this postulate does not follow neatly from the current state of the evidence, and the rhetorical language used risks negatively impacting patients by implying that these symptoms are ‘attention seeking’ behaviours. In this response, written by a group of Tourette syndrome researchers, clinicians, and individuals with tics, we detail concerns with the paper.

Journal ArticleDOI
TL;DR: This paper reviewed and summarized the literature relevant to distinguish the two, with recommendations to clinicians for diagnosis and management, highlighting areas for future emphasis and research, and highlighted areas to be highlighted.
Abstract: Over the past 3 years, a global phenomenon has emerged characterized by the sudden onset and frequently rapid escalation of tics and tic-like movements and phonations. These symptoms have occurred not only in youth known to have tics or Tourette syndrome (TS), but also, and more notably, in youth with no prior history of tics. The Tourette Association of America (TAA) convened an international, multidisciplinary working group to better understand this apparent presentation of functional neurological disorder (FND) and its relationship to TS. Here, we review and summarize the literature relevant to distinguish the two, with recommendations to clinicians for diagnosis and management. Finally, we highlight areas for future emphasis and research.

Journal ArticleDOI
TL;DR: In this article, the authors classified ten mental disorders by genomic structural equation modeling (SEM), which leverages summary results of genome-wide association studies, and found that they are genetically inter-correlated with one another and can be separated based on their general psychopathology.
Abstract: Typically, mental disorders are defined and classified based on clinical symptoms and syndromes. Although clinically useful, current diagnostic systems for psychiatry cause concerns due to the lack of biological mechanisms. Deciphering the relationships among psychiatric traits according to their genetic basis may facilitate understanding the biological mechanisms of psychiatric disorders. Ten mental disorders were classified by genomic structural equation modeling (SEM), which leverages summary results of genome-wide association studies. Attention-deficit/hyperactivity disorder (ADHD), anorexia nervosa (AN), anxiety disorder (ANX), autism spectrum disorder (ASD), bipolar disorder (BD), major depressive disorder (MDD), obsessive-compulsive disorder (OCD), posttraumatic stress disorder (PTSD), schizophrenia (SZ), and Tourette syndrome (TS) were included. The analysis indicates that they are genetically inter-correlated with one another and can be separated based on their general psychopathology. Most disorders have a close partner, forming pairs of traits; only TS is a relatively distinctive condition. At a higher level, MDD, ANX, ADHD, ASD, and PTSD cluster together, while OCD, AN, and TS cluster together. Together, the ten traits constitute a hierarchical classificatory system. This study allows inference of genetically determined classification of the ten mental disorders, which may biologically inform the current diagnostic framework and treatment regimens for mental disorders.

Journal ArticleDOI
TL;DR: In this article , the authors explored the role of self-esteem in mediating the relationship between psychosocial stress and social adjustment among adolescents with Tourette syndrome, and found that without comorbidities, the selfesteem of adolescents with tics was more pronounced than those with comorebidities.

Journal ArticleDOI
TL;DR: Gilles de la Tourette syndrome (TS) is a common, childhood-onset psychiatric disorder characterized by persistent motor and vocal tics as mentioned in this paper , which is a heterogeneous disorder in which the phenotypic expression may be affected by environmental factors, such as immune responses.
Abstract: Gilles de la Tourette syndrome (TS) is a common, childhood-onset psychiatric disorder characterized by persistent motor and vocal tics. It is a heterogeneous disorder in which the phenotypic expression may be affected by environmental factors, such as immune responses. Furthermore, several studies have shown that genetic factors play a vital role in the etiology of TS, as well as its comorbidity with other disorders, including attention deficit hyperactivity disorder, obsessive-compulsive disorder, and autism spectrum disorder. TS has a complex inheritance pattern and, according to various genetic studies, several genes and loci have been correlated with TS. Genome-wide linkage studies have identified Slit and Trk-like 1 (SLITRK1) and histidine decarboxylase (HDC) genes, and candidate gene association studies have extensively investigated the dopamine and serotonin system genes, but there have been no consistent results. Moreover, genome-wide association studies have implicated several genetic loci; however, larger study cohorts are needed to confirm this. Copy number variations, which are polymorphisms in the number of gene copies due to chromosomal deletions or duplications, are considered another significant source of mutations in TS. In the last decade, whole genome/exome sequencing has identified several novel genetic mutations in patients with TS. In conclusion, more studies are needed to reveal the exact mechanisms of underlying TS, which may help to provide more information on the prognosis and therapeutic plans for TS.

Journal ArticleDOI
TL;DR: Both internet-delivered ERP and structured education were associated with improvements in tic severity, but ERP led to higher treatment response rates.
Abstract: Key Points Question Is therapist-supported, internet-delivered exposure and response prevention (ERP) efficacious and cost-effective for young people with Tourette syndrome or chronic tic disorder? Findings In this randomized clinical trial of 221 youths with Tourette syndrome or chronic tic disorder who received either therapist-supported internet-delivered ERP or structured education, both groups significantly improved over time, with no between-group differences in tic severity. However, ERP was associated with significantly higher treatment response rates (47% vs 29%) at little additional cost. Meaning Both internet-delivered ERP and structured education were associated with improvements in tic severity, but ERP led to higher treatment response rates.

Journal ArticleDOI
TL;DR: Embedded closed-loop deep DBS was feasible, safe, and had a comparable outcome to conventional TS DBS for the treatment of tics in a single-center double-blinded safety and feasibility trial.
Abstract: Importance Because Tourette syndrome (TS) is a paroxysmal disorder, symptomatic relief in individuals with TS may be possible through the application of stimulation only during the manifestation of human tic neural signatures. This technique could be capable of suppressing both motor and vocal tics and would have similar effectiveness to conventional continuous deep brain stimulation (DBS). Objective To evaluate the feasibility, safety, and clinical effectiveness of bilateral centromedian-parafascicular complex thalamic closed-loop DBS as a treatment for medication-refractory TS. Design, Setting, and Participants This single-center double-blinded safety and feasibility trial was conducted between February 2014 and June 2020. Six individuals with TS were screened and recruited from the Norman Fixel Institute at the University of Florida. The primary outcome was measured at 6 months, and participants were followed up for the duration of the neurostimulator battery life. Independent ratings that compared closed-loop and conventional DBS were videotaped. The first 2 of 6 individuals with TS were excluded from the study because the technology for embedded closed-loop capability was not yet available. The date of analysis was August 2020. Interventions DBS therapy controlled by an embedded closed-loop stimulation system. Main Outcomes and Measures The primary clinical outcome measure was a minimum of a 40% reduction in the YGTSS score at 6 months following DBS. There was also a comparison of conventional DBS with closed-loop DBS using the Modified Rush Videotape Rating Scale for Tic. Results The mean (SD) age at TS diagnosis for the cohort was 8.5 (2.9), and the mean (SD) disease duration was 23.7 (5.8) years. Four individuals with TS were analyzed (2 male, 2 female; mean [SD] age, 23.7 [5.8] years). The study showed the closed-loop approach was both feasible and safe. One of the novelties of this study was that a patient-specific closed-loop paradigm was created for each participant. The features and stimulation transition speed were customized based on the signal quality and the tolerance to adverse reactions. The mean (SD) therapeutic outcome with conventional DBS was 33.3% (35.7%) improvement on the YGTSS and 52.8% (21.9%) improvement on the Modified Rush Videotape Rating Scale. Two of 4 participants had a primary outcome variable improvement of 40% meeting the primary efficacy target. When comparing closed-loop DBS with conventional DBS using a Wilcoxon sign-rank test, there was no statistical difference between tic severity score and both approaches revealed a lower tic severity score compared with baseline. The study was feasible in all 4 participants, and there were 25 total reported adverse events with 3 study-related events (12%). The most common adverse events were headache and anxiety. Conclusions and Relevance Embedded closed-loop deep DBS was feasible, safe, and had a comparable outcome to conventional TS DBS for the treatment of tics. Trial Registration ClinicalTrials.gov Identifier: NCT02056873.

Journal ArticleDOI
TL;DR: MC treatment demonstrates good efficacy and tolerability in adult GTS patients and Predilection for smoking rather than using oil drops requires further comparative studies to evaluate the efficacy of each.
Abstract: Objectives Assessing the effectiveness and tolerability of medical cannabis (MC) treatment on Gilles de la Tourette syndrome (GTS) patients. Methods We report on an open-label, prospective study on the effect of MC on adult GTS patients. MC mode of use was decided by the treating neurologist and the patient. Δ9-Tetrahydrocannabinol (Δ9-THC) and cannabidiol (CBD) content within MC product and monthly dose were titrated during the study. Following treatment initiation, patients were assessed after 4 and 12 weeks for efficacy, tolerability, and side effects. Results Eighteen patients entered the study. Baseline Yale Global Tic Severity Scale- (YGTSS) Total (range 0-100) was 60.3 ± 17.1. Three patients did not reach the end of follow-up period. The most common mode of administration was smoking (80%). Following twelve weeks of treatment, a significant 38% average reduction (p = 0.002) of YGTSS-Total and a 20% reduction (p = 0.043) of Premonitory Urge for Tic Scale (PUTS) were observed. Common side effects were dry mouth (66.7%), fatigue (53.3%), and dizziness (46.7%). Three patients suffered from psychiatric side effects including worsening of obsessive compulsive disorder (stopped treatment), panic attack, and anxiety (resolved with treatment modification). Six patients (40%) reported cognitive side effects regarding time perception, visuospatial disorientation, confusion, slow processing speed, and attention. Conclusions MC treatment demonstrates good efficacy and tolerability in adult GTS patients. Predilection for smoking rather than using oil drops requires further comparative studies to evaluate the efficacy of each. Cognitive and psychiatric side effects have to be monitored and addressed.

Journal ArticleDOI
01 Feb 2022-Cortex
TL;DR: In this article , a Go/NoGo task was performed on children with Tourette syndrome (TS) and attention-deficit/hyperactivity disorder (ADHD) during EEG recordings.

Journal ArticleDOI
TL;DR: In this article , the authors used a neuro-computational model of hierarchically organized cortico-basal ganglia-thalamo-cortical loops to shed more light on habit formation and its alteration in Tourette patients.
Abstract: Devaluation protocols reveal that Tourette patients show an increased propensity to habitual behaviors as they continue to respond to devalued outcomes in a cognitive stimulus-response-outcome association task. We use a neuro-computational model of hierarchically organized cortico-basal ganglia-thalamo-cortical loops to shed more light on habit formation and its alteration in Tourette patients. In our model, habitual behavior emerges from cortico-thalamic shortcut connections, where enhanced habit formation can be linked to faster plasticity in the shortcut or to a stronger feedback from the shortcut to the basal ganglia. We explore two major hypotheses of Tourette pathophysiology-local striatal disinhibition and increased dopaminergic modulation of striatal medium spiny neurons-as causes for altered shortcut activation. Both model changes altered shortcut functioning and resulted in higher rates of responses towards devalued outcomes, similar to what is observed in Tourette patients. We recommend future experimental neuroscientific studies to locate shortcuts between cortico-basal ganglia-thalamo-cortical loops in the human brain and study their potential role in health and disease.

Journal ArticleDOI
TL;DR: Online remote behavior therapy is a promising tool for behavioral therapies for patients with Tourette Syndrome and may represents an alternative treatment option.
Abstract: Objective To evaluate the clinical effectiveness of online remote behavior therapy, compared with face-to-face therapy in reducing tics and co-occurring disorders associated with the tics in a sample of youths with Tourette Syndrome. Design A randomized controlled trial. TS patients were randomized to receive face-to-face or online remote behavior therapy. Participants 40 children aged between 9 and 16 years affected by Tourette Syndrome. Results Online remote and face-to-face behavior therapy are equally effective in the treatment of tics and co-occurring disorders in children and adolescents affected by Tourette Syndrome. Both groups showed an improvement in the severity of tics, obsessive-compulsive symptoms, and anxiety symptoms, as assessed by neuropsychological findings. Online remote behavior therapy was more effective for reducing depressive symptoms than face-to-face behavior therapy. Conclusions Online remote behavior therapy is a promising tool for behavioral therapies for patients with Tourette Syndrome and may represents an alternative treatment option.

Journal ArticleDOI
TL;DR: In this paper , the authors examine the treatments currently available for Tourette syndrome (TS) and discuss evolving therapies, spanning behavioral, pharmacologic, complementary and alternative medicine, and neuromodulation approaches.
Abstract: This study aims to examine the treatments currently available for Tourette syndrome (TS) and to discuss evolving therapies, spanning behavioral, pharmacologic, complementary and alternative medicine, and neuromodulation approaches.Behavioral therapies have undergone several modifications to improve accessibility, including transitioning to a virtual format which is particularly important in the current pandemic. There are several recent or ongoing pharmacologic studies that have shown promise including the selective D1 receptor antagonist ecopipam and various cannabinoid compounds. Adaptive DBS may enable the physiologic markers of tics to determine stimulation parameters and improve tic outcomes related to neuromodulation. In recent years, there has been a wealth of research across multiple treatment domains in the TS field. This review highlights exciting and new potential options for the future treatment of patients with TS.

Journal ArticleDOI
TL;DR: A systematic literature search was conducted in seven major databases with various keywords relating to closed-loop DBS, for non-randomised cohort studies, finalised clinical trials, case reports, and nonrandomised control trials published between 2011 and 2021 as discussed by the authors .

Journal ArticleDOI
TL;DR: The strong association between PU and tic location further supports the hypothesis that PUs represent the core of PTD, and future therapies should focus on treating PUs to achieve greater tic reduction.
Abstract: Objective In patients with Tourette syndrome and other primary tic disorders (PTDs), tics are typically preceded by premonitory urges (PUs). To date, only a few studies have investigated the location and frequency of PUs, and contrary to clinical experience, the results suggest that PUs are not located in the same anatomic region as the tics. This study aimed to further explore PU location and frequency in detail, differentiating the kind and complexity of the corresponding tics, in a large sample of patients with PTD. Methods A total of 291 adult (≥ 18 years) patients with a confirmed diagnosis of chronic PTD were included. The study was conducted online, assement included tics and the general characterization of PUs and a sophisticated body drawing for locating PUs. Results We found that PUs were located in the same body area as, or in direct proximity to, the corresponding tic. Most frequently, PUs were located in the face and at the head (62.1%). Compared with simple tics, complex (motor and vocal) tics were more often preceded by a PU; but there was no difference in PU frequency observed between motor tics and vocal tics. PUs were more often experienced at the front than at the back of the body (73% vs. 27%), while there was no difference between the right and left sides (41.6% vs. 41.3%). Conclusion The strong association between PU and tic location further supports the hypothesis that PUs represent the core of PTD. Accordingly, future therapies should focus on treating PUs to achieve greater tic reduction.

Journal ArticleDOI
TL;DR: In this paper, a cross-sectional study aimed to determine whether Mycoplasma pneumoniae (M. pneumoniae) IgG positivity is associated with the presence or severity of chronic tic disorders.
Abstract: Infectious pathogens may represent an environmental risk factor for chronic tic disorders (CTD). This cross-sectional study aimed to determine whether Mycoplasma pneumoniae (M. pneumoniae) IgG positivity is associated with the presence or severity of tics. We compared M. pneumoniae IgG positivity across three groups: children and adolescents (3–16 years) with CTD (CTD group; n = 302); siblings (3–10 years) of people with CTD who developed tics within a seven-year follow-up period (tic onset group; n = 51); siblings (4–10 years) who did not develop tics within the study period and were ≥10-years-old at their last assessment (unaffected group; n = 88). The relationship between M. pneumoniae IgG positivity and the presence and severity of tics was analysed using multilevel models controlling for site, family relatedness, sex, age, presence of comorbid obsessive–compulsive and/or attention-deficit/hyperactivity disorder and use of psychotropic medication. M. pneumoniae IgG positivity was not associated with the presence of CTD, or the first onset of tics as compared to siblings who remained unaffected. M. pneumoniae IgG positivity was associated with a higher tic severity score within the CTD group (β = 2.64, s.e. = 1.15, p = 0.02). It is possible that M. pneumoniae infection influences tic severity in CTD or, that having more severe tics, increases the risk of infection. However, it is more likely that the association observed in this study reflects a propensity toward enhanced immune responses in people with CTD and that, rather than a causal relationship, infection and greater tic severity are indirectly linked via shared underlying immune mechanisms.

Journal ArticleDOI
01 Feb 2022-Cureus
TL;DR: The treatment of Tourette's syndrome can be broadly classified into non-pharmacological and pharmacological treatment as mentioned in this paper , which includes various behavioural interventions that can be helpful in situations when patients are tolerant of medical treatments.
Abstract: Tourette's Syndrome (TS), in which patients have sudden, repeated, involuntary twitches and movements, called tics, is a condition of the nervous system. They can be motor, vocal, simple, or complex tics. It can be physically, emotionally, mentally, and socially distressing and challenging for those suffering from it. Usually, it is accompanied by various comorbidities like attention-deficit hyperactivity disorder, obsessive-compulsive disorder, and sleep disorders. A variety of environmental and genetic factors are also associated with tics in TS like the first-degree relatives are more at risk of developing TS.TS is heterogeneous with complicated patterns of inheritance and phenotypic manifestations. There is a strong association between common single nucleotide polymorphisms (SNP, s) in the SLITRK1 gene and TS. Environmental factors like prenatal, postnatal, and perinatal factors directly influence tics in TS. These factors are low birth weight, intrauterine growth retardation (IGR), and various infections. The treatment of TS can be broadly classified into non-pharmacological and pharmacological treatment. Non-pharmacological therapy includes various behavioural interventions that can be helpful in situations when patients are tolerant of medical treatments. Psychoeducation and counselling play an essential role in the treatment of TS. It is vital to give a proper understanding to the patient and their family about the disease. Cognitive-behavioral intervention for tics, cognitive-behavioral therapy, exposure and response prevention, relaxation techniques, deep brain stimulation, and habit reversal training are the commonly used therapies for tics. These therapies have shown good efficacy because it improves the Yale Global Tic Severity Scale score (YGTSS) significantly. And they show effectiveness in patients who are irresponsive to medical treatment. The main lines of medical treatment are antipsychotics and alpha agonists. Typical (haloperidol, pimozide) or atypical (aripiprazole, risperidone, olanzapine) Antipsychotics differ in their side effects, efficacy, and tolerance in different age groups of children. Haloperidol was the first drug approved by the Food and Drug Administration for tics, but later on, new developments and improvements were made as far as drug therapy is concerned. The alpha-agonist most commonly used is clonidine which is also available in the form of adhesive patches. Another alpha agonist which is also widely used is guanfacine. Botulinum toxin and baclofen have also shown efficacy in dealing with tics in TS with other comorbidities. We will review in this article all the main lines of treatment and their effectiveness in TS.

Journal ArticleDOI
Ming Lai, Yuanyuan Li, Dan Luo, Jiajun Xu, Jing Li 
TL;DR: In this paper , a 13-year-old boy with a history of Tourette syndrome presented with acute tongue-biting without positive neurologic examination or auxiliary examination results, except for a weakly positive finding for D2R antibodies in the serum sample.
Abstract: Tourette syndrome (TS) is a neuropsychiatric disorder characterized by repetitive and patterned tics. Its onset correlates with dysfunctions in immunological activation and neurotransmitters. Autoimmune movement disorders such as dopamine-2 receptor antibody encephalitis (D2R encephalitis) may go undiagnosed in TS patients seeking medical help for tic symptoms only. Here, we present a clinical case of D2R encephalitis in a TS patient.A 13-year-old boy with a history of TS presented with acute tongue-biting without positive neurologic examination or auxiliary examination results, except for a weakly positive finding for D2R antibodies in the serum sample. He was initially diagnosed with possible D2R encephalitis, but the influence of TS could not be ruled out. In addition to psychotropics, we administered immunotherapy early based on clinical characteristics, and his symptoms were ameliorated significantly. During the follow-up, he was diagnosed with definite D2R encephalitis, and the dosage of psychotropics was further adjusted for fluctuating symptoms.Our case suggests that clinicians should discern D2R encephalitis in TS patients when tics are the primary symptoms. Administering immunotherapy early, according to clinical characteristics, may benefit the patient. Moreover, the features of premonitory urges could help evaluate the state of TS.

Journal ArticleDOI
TL;DR: In this paper , the authors evaluated the relationship between the anatomical location of stimulation fields and clinical responses, including therapeutic and side effects, and suggested that meticulous programming with careful monitoring of clinical symptoms may improve outcomes.
Abstract: Deep brain stimulation (DBS) of the centromedian thalamic nucleus has been reportedly used to treat severe Tourette syndrome, yielding promising outcomes. However, it remains unclear how DBS electrode position and stimulation parameters modulate the specific area and related networks. The authors aimed to evaluate the relationships between the anatomical location of stimulation fields and clinical responses, including therapeutic and side effects.The authors collected data from 8 patients with Tourette syndrome who were treated with DBS. The authors selected the active contact following threshold tests of acute side effects and gradually increased the stimulation intensity within the therapeutic window such that acute and chronic side effects could be avoided at each programming session. The patients were carefully interviewed, and stimulation-induced side effects were recorded. Clinical outcomes were evaluated using the Yale Global Tic Severity Scale, the Yale-Brown Obsessive-Compulsive Scale, and the Hamilton Depression Rating Scale. The DBS lead location was evaluated in the normalized brain space by using a 3D atlas. The volume of tissue activated was determined, and the associated normative connective analyses were performed to link the stimulation field with the therapeutic and side effects.The mean follow-up period was 10.9 ± 3.9 months. All clinical scales showed significant improvement. Whereas the volume of tissue activated associated with therapeutic effects covers the centromedian and ventrolateral nuclei and showed an association with motor networks, those associated with paresthesia and dizziness were associated with stimulation of the ventralis caudalis and red nucleus, respectively. Depressed mood was associated with the spread of stimulation current to the mediodorsal nucleus and showed an association with limbic networks.This study addresses the importance of accurate implantation of DBS electrodes for obtaining standardized clinical outcomes and suggests that meticulous programming with careful monitoring of clinical symptoms may improve outcomes.

Journal ArticleDOI
TL;DR: In this article , the authors evaluated the efficacy of relaxation training alone or alongside other CBIT components for reducing tic severity in patients with Tourette's disorder and found that relaxation training is effective when included in conjunction with CBIT.