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Individuals living with Tourette syndrome (TS) may benefit from cognitive assessment, as it may elucidate specific difficulties for which someone could receive accommodation and highlight relative strengths to build upon. Eventually, cognitive assessment could help predicting symptom trajectory or treatment outcome. However, the literature on cognition in TS is often equivocal, which may be at least partly attributed to small samples, inconsistent consideration of co-occurring conditions, psychotropic medication, and developmental influence. Here, we aimed to provide a comprehensive overview of the literature on cognition in TS, to disentangle conflicting findings and make suggestions of how the findings can be applied to promote better outcomes., Competing Interests: Disclosure The authors have no conflict of interest to disclose., (Copyright © 2024 Elsevier Inc. All rights reserved.)

Objectives: Recent survey data suggest that a high proportion of patients with Tourette syndrome (TS) experience pain, yet pain features in TS have not been previously investigated in a systematic manner. This article reviews the current understanding and impact of pain in TS as well as identifies possible areas for emphasis for future research on pain in TS. Methods: Using a comprehensive search strategy in two relevant research databases (PubMed and Scopus), we searched for relevant peer-reviewed, primary research articles, and review articles. Search terms used were Tourette syndrome, tic disorder, pain, pain management, sensory, and sensory gating. Results: A total of 116 pertinent articles were identified. Pain is reported by 47%-60% of individuals with TS and may relate to different aspects of tic phenomenology or other causes. Pain is more prevalent among TS patients than in the general population and negatively impacts quality of life. To standardize future research efforts, we propose the following classification: tic-related immediate pain, tic-related delayed injury/pain, suppression-related pain, premonitory urge-related pain, and associated primary pain syndromes. Altered sensory gating and interoceptive processing abnormalities are possible mechanisms contributing to pain in TS but warrant further study. Despite pain prevalence, most TS clinical rating scales and outcome measures used in therapeutic studies do not incorporate sufficient information regarding pain. Therapies known to improve pain in non-TS conditions that are also reported to improve tics have not been investigated for their effects on pain among TS patients. Conclusion: TS can be associated with a chronic pain syndrome that negatively affects quality of life. Future research using a systematic framework is needed to better understand pain cause(s) and prevalence, develop appropriate assessment methods, establish outcome measures, and understand mechanisms of pain in TS. Such investigations are likely to lead to therapeutic options for this troublesome symptom.

Background: Tics are the hallmark of Tourette syndrome (TS) and chronic tic disorders (CTD). Although typically involving the face, especially at onset, tics may involve any muscle under voluntary control, including axial muscles of the neck (causing head movements), shoulders and trunk (thorax and abdomen). We aimed to characterize these tics and provide a clinical frame for their associations and complications., Materials and Methods: We reviewed video recordings and clinical history of 196 patients with TS or CTD according to DSM-5., Results: Any axial tic was identified in 75% of patients. Tic distribution were head (n = 113, 57.6%), shoulder (n = 91, 46.4%), and trunk (n = 63, 32.2%). There were no differences in sex, age at onset or at evaluation between patients with and without axial tics. The most common axial tics by anatomical distribution were head turning, bilateral synchronous shoulder elevation and trunk jerks; however, tic phenomenology was quite variable. A greater severity of tics (P = 0.018) was associated with axial tics in the multivariate regression analysis. Head/neck tics associated with simple phonic tics (P = 0.002); whereas shoulder and trunk tics associated with complex motor tics (P < 0.05) in a bivariate analysis. Neck pain, breathing interference, sleep limitation and radiculopathy, secondary to axial tics were complications observed in a proportion of these cases., Conclusions: Axial tics are commonly observed in patients with TS/CTD with variable phenomenology. They associate with greater tic severity, phonic tics and complex motor tics. They may result in neck pain, breathing interference, sleeping problems and cervical spine injuries., Competing Interests: Declarations Conflicts of interest On behalf of all the authors, the corresponding author states that there is no conflict of interest., (© 2024. Springer-Verlag GmbH Germany, part of Springer Nature.)

Background: Despite research demonstrating sleep disturbance in children with Tourette syndrome (TS), few studies have examined bedtime regularity and sleep sufficiency, two important sleep health dimensions. Therefore, this study examined bedtime regularity and sleep sufficiency in children with TS relative to matched healthy control subjects, and its associated demographic, clinical, and behavioral factors., Methods: Participants were 384 parents or caregivers of children aged three to 17 years, including 192 with current TS and 192 matched healthy control subjects drawn from the 2020-2021 cycle of the National Survey of Children's Health. Parents completed questions assessing demographic (i.e., age, race, sex), clinical (i.e., attention-deficit/hyperactivity disorder [ADHD], autism spectrum disorder, anxiety, depression, tic severity, behavioral or conduct problems, ADHD medication, health condition-related impairment), and behavioral (i.e., screen time) characteristics. Mann-Whitney U test and chi-square test of independence were performed to compare groups on bedtime regularity and sleep sufficiency, respectively. Ordinal regression and binary logistic regression without and with backward elimination were performed to evaluate indicators of bedtime regularity and sleep sufficiency, respectively, in children with TS., Results: Children with current TS had significantly poorer bedtime regularity, but not sleep sufficiency, relative to matched healthy control subjects. In children with TS, anxiety and two or more hours of daily screen time were associated with higher likelihood of poor bedtime regularity. Autism was associated with lower likelihood of insufficient sleep, and depression was associated with increased likelihood of insufficient sleep., Conclusions: Findings put forth screen time, anxiety, and depression as intervention targets to optimize sleep health in children with TS., Competing Interests: Declaration of competing interest Ms. Swisher has received research funding from the American Academy of Sleep Medicine and the National Science Foundation. Dr. Ricketts has received honoraria and funding from the Tourette Association of America, the National Institute of Mental Health, and Brain and Behavior Research Foundation. She has received travel support and funding from the American Academy of Sleep Medicine Foundation and honoraria from the Centers for Disease Control and Prevention, Springer Nature, and Wink Sleep. Ms. Liu declares no conflicts of interest., (Copyright © 2024 Elsevier Inc. All rights reserved.)

Background: Klinefelter syndrome (47, XXY) is the most common sex chromosome aneuploidy. In addition to male hypergonadotropic hypogonadism, a wide range of neurodevelopmental disorders, anxiety and affective symptoms have been reported in a substantial proportion of cases., Case Description: We document the rare case of a 43-year-old man diagnosed with Klinefelter syndrome and co-morbid Gilles de la Tourette syndrome. He presented with multiple motor and vocal tics since adolescence, as well as anxiety and affective symptoms as his main tic-exacerbating factors. Tic severity was rated as marked (Yale Global Tic Severity Scale score of 78/100), and recommendations for the treatment of both tics and psychiatric co-morbidities were formulated., Discussion: Neurodevelopmental tics in the context of Klinefelter syndrome have been previously documented in three cases only. Gilles de la Tourette syndrome is 3-4 times more common in males than females and its etiological factors include multiple genetic components (genetic heterogeneity). Our case report widens the spectrum of neurodevelopmental disorders observed in the context of Klinefelter syndrome and contributes to genetic research on the role of the X chromosome in the pathophysiology of tic disorders., (© 2024. The Author(s).)

Purpose: The purpose of this study was to report a case of Tourette syndrome, exacerbated in adulthood, that was diagnosed after bilateral lens luxation and recurrent retinal detachment due to self-injury., Methods: This was a case report., Results: A 35-year-old man presented with a sudden abnormality in vision and bilateral lens luxation. The patient successfully underwent bilateral lens extraction and intrascleral intraocular lens fixation; however, vitreous hemorrhage and retinal detachment occurred in the left eye. The retinal detachment was caused by a giant retinal tear and retinal dialysis. Vitrectomy was performed. However, retinal detachment recurred with proliferative vitreoretinopathy. Retinal detachment subsequently occurred in the right eye. Self-injury to the eye was observed before surgical treatment. The patient was consequently diagnosed with Tourette syndrome., Conclusion: Tourette syndrome, which may be accompanied by self-injurious behavior, is a disorder that typically develops in childhood but rarely exacerbates during adulthood. A diagnosis of Tourette syndrome should be considered in cases of unexplained retinal detachment with traumatic features.

Tourette syndrome (TS) is a complex neurodevelopmental disorder characterized by the presence of tics, frequently accompanied by a variety of neuropsychiatric comorbidities. A subset of patients with TS present with severe and disabling symptoms, requiring prompt therapeutic intervention. Some of these manifestations may result in medical emergencies when severe motor or phonic tics lead to damage of anatomical structures closely related to the tic. Examples include myelopathy or radiculopathy following severe neck ("whiplash") jerks or a variety of self-inflicted injuries. In addition to self-aggression or, less commonly, allo-aggression, some patients exhibit highly inappropriate behavior, suicidal tendencies, and rage attacks which increase the burden of the disease and are important components of "malignant TS". This subset of TS is frequently associated with comorbid obsessive-compulsive disorder. Therapeutic measures include intensive behavioral therapy, optimization of oral pharmacotherapy, botulinum toxin injections, and deep brain stimulation., Competing Interests: Declarations Informed consent for video/photography and publication in a scientific journal was provided by patients or legal proxy, approved by the Research Committee of Baylor College of Medicine., (Copyright © 2024 Elsevier Ltd. All rights reserved.)

Across a range of neurodevelopmental conditions, a diagnosis of attention deficit hyperactivity disorder (ADHD) has been shown to be associated with executive function impairment. However, the DSM-V emphasis upon viewing psychological characteristics as existing on a continuous distributed quantitative dimension has enabled the opportunity to consider the influence of sub-diagnostic or sub-referral levels of these psychological characteristics upon cognitive function. This study adopted a continuum approach to the consideration of this ADHD influence and examined the extent to which the difference in parental reported executive functions between children with Tourette syndrome (TS) or typically developing children could be mediated by a concurrent group difference in the possession of sub-referral levels of ADHD-like characteristics. A total of 146 children, 58 with reported TS diagnosis, participated. Parental report measures of ecological executive functioning, the Child Executive Functioning Inventory, and the Vanderbilt ADHD Diagnostic Parent Rating Scale were employed. The analyses with a full sample and a sub-referral sample revealed significant group differences in most of the key measures. In addition, these measures were highly correlated even when controlling for age and gender. A series of mediation analyses indicated that in all models, the ADHD-like measures significantly mediated the group difference in executive function. These results suggest that sub-referrals levels of ADHD-like characteristics continue to contribute to executive challenges in TS. Future intervention research targeting these executive functions should consider the presence of ADHD-like characteristics at sub-referral levels of possession.

To report the results of 'responsive' deep brain stimulation (DBS) for Tourette syndrome (TS) in a National Institutes of Health funded experimental cohort. The use of 'brain derived physiology' as a method to trigger DBS devices to deliver trains of electrical stimulation is a proposed approach to address the paroxysmal motor and vocal tic symptoms which appear as part of TS. Ten subjects underwent bilateral staged DBS surgery and each was implanted with bilateral centromedian thalamic (CM) region DBS leads and bilateral M1 region cortical strips. A series of identical experiments and data collections were conducted on three groups of consecutively recruited subjects. Group 1 (n = 2) underwent acute responsive DBS using deep and superficial leads. Group 2 (n = 4) underwent chronic responsive DBS using deep and superficial leads. Group 3 (n = 4) underwent responsive DBS using only the deep leads. The primary outcome measure for each of the 8 subjects with chronic responsive DBS was calculated as the pre-operative baseline Yale Global Tic Severity Scale (YGTSS) motor subscore compared to the 6 month embedded responsive DBS setting. A responder for the study was defined as any subject manifesting a ≥ 30 points improvement on the YGTSS motor subscale. The videotaped Modified Rush Tic Rating Scale (MRVTRS) was a secondary outcome. Outcomes were collected at 6 months across three different device states: no stimulation, conventional open-loop stimulation, and embedded responsive stimulation. The experience programming each of the groups and the methods applied for programming were captured. There were 10 medication refractory TS subjects enrolled in the study (5 male and 5 female) and 4/8 (50%) in the chronic responsive eligible cohort met the primary outcome manifesting a reduction of the YGTSS motor scale of ≥ 30% when on responsive DBS settings. Proof of concept for the use of responsive stimulation was observed in all three groups (acute responsive, cortically triggered and deep DBS leads only). The responsive approach was safe and well tolerated. TS power spectral changes associated with tics occurred consistently in the low frequency 2-10 Hz delta-theta-low alpha oscillation range. The study highlighted the variety of programming strategies which were employed to achieve responsive DBS and those used to overcome stimulation induced artifacts. Proof of concept was also established for a single DBS lead triggering bi-hemispheric delivery of therapeutic stimulation. Responsive DBS was applied to treat TS related motor and vocal tics through the application of three different experimental paradigms. The approach was safe and effective in a subset of individuals. The use of different devices in this study was not aimed at making between device comparisons, but rather, the study was adapted to the current state of the art in technology. Overall, four of the chronic responsive eligible subjects met the primary outcome variable for clinical effectiveness. Cortical physiology was used to trigger responsive DBS when therapy was limited by stimulation induced artifacts., (© 2024. The Author(s).)

Purpose of Review: Tourette syndrome (TS) and chronic tic disorders, collectively referred to as TS, are relatively common childhood onset neuropsychiatric conditions associated with functional impairment and distress. Over the past several years, clinical research has contributed to the advancement of the field's understanding of mechanisms and clinical correlates of TS. This progress has led to the development of key assessment tools and the implementation of novel interventions for individuals with TS., Recent Findings: This article provides a review of innovative TS research focusing on four key themes: investigation of clinical phenomenology of TS; validation of assessment tools for TS; dissemination of current evidence-based treatments for TS; and exploration of new intervention programs., Summary: Cumulatively, this growing body of work presents considerable progress and provides a path forward to improve the assessment and treatment of TS., (Copyright © 2023 Wolters Kluwer Health, Inc. All rights reserved.)

Background: Tics and stereotypies are childhood-onset repetitive behaviours that can pose significant diagnostic challenges in clinical practice. Both tics and stereotypies are characterised by a complex co-morbidity profile, however little is known about the co-occurrence of these hyperkinetic disorders in the same patient population., Objective: This review aimed to assess the relationship between tics and stereotypies when these conditions present in co-morbidity., Methods: We conducted a systematic literature review of original studies on co-morbid tics and stereotypies, according to the standards outlined in the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines., Results: Our literature search identified six studies of suitable sample size (n ≥ 40) presenting data on the association between tics and stereotypies in otherwise typically developing patients. A considerable proportion (23%) of patients diagnosed with stereotypic movement disorder present with co-morbid tics (range 18-43%). Likewise, the prevalence of primary stereotypies is increased in patients with tic disorders such as Tourette syndrome (8%, range 6-12%)., Discussion: Tics and stereotypies can often develop in co-morbidity. The association of tics and stereotypies in the same patient has practical implications, in consideration of the different treatment approaches. Future research should focus on the assessment and management of both conditions, particularly in special populations (e.g. patients with pervasive developmental disorders)., (© 2023. The Author(s).)

There is increasing recognition of the high prevalence of sleep issues in children with Tourette syndrome (TS), a condition characterised by motor and vocal tics. Overnight polysomnography (PSG) has been the primary mode of sleep assessment in the TS literature, despite the extensive use of actigraphy in other neurodevelopmental populations. As a result, there are existing research gaps surrounding day-to-day variability of sleep in TS and links to daytime functioning. This study adopts a naturalistic, intensive longitudinal design to examine sleep in children with TS while considering potential links to tic severity and daytime functioning. Participants were 34 children aged between 8 and 12 years (12 with TS, 22 neurotypical controls). Wrist actigraphs tracked sleep-wake cycles across two weeks and a battery of scales and cognitive assessments measured sleep disturbances and daytime functioning. Mixed models using N = 476 nights of actigraphy data found that relative to controls, children with TS had significantly increased time in bed, increased sleep onset latency, reduced sleep efficiency, lower subjective sleep quality, but comparable actual sleep time. Higher self-report tic severity at bedtime did not predict increased sleep onset latency. In the sleep disturbance scale, 83.33 % of children with TS met the clinical cut-off for a sleep disorder. Parent-report emotional, behavioural, and executive difficulties were greater in the TS group relative to controls, but performance on cognitive tasks was comparable between groups. Together, findings highlight sleep disturbances as an important clinical factor to consider in the management of TS, though further research is required to substantiate findings in larger-scale studies. This study demonstrates the feasibility of assessing sleep via actigraphy in children with TS, supporting more widespread use in the future., Competing Interests: Declaration of competing interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2023 The Authors. Published by Elsevier B.V. All rights reserved.)

Tourette syndrome is characterized by at least two motor tics and one vocal tic, which persist for over a year. Infrequently, tics can manifest as blocking tics in speech when they prevent a person from starting to speak or interrupt their speech flow. Vocal blocking tics (VBTs) resemble stuttering, and they can be difficult to differentiate from each other. A previous report described two patients with severe VBTs who did not benefit from stuttering-therapy-based speech therapy and were treated effectively with cannabis-based medicine. Here, we present the cases of two patients, seven- and nine-year-old boys, who benefited from speech therapy in which stuttering therapy techniques were used. Detailed descriptions of the interventions are included. Further research is needed to test the effectiveness of speech therapy in treating VBTs in a larger group of children with Tourette syndrome., Competing Interests: Declaration of conflicting interestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

There is an increasing need to identify and treat sleep disturbances in Tourette syndrome (TS), a neurodevelopmental condition characterized by tics. This study explored sleep, tics, and executive functioning in children with TS (n=136) and neurotypical controls (n=101) through parent-report scales and open-ended questions. 85% of children with TS scored in the clinical range for a sleep disorder. Higher tic severity predicted increased sleep disturbances and executive difficulties. Qualitative insights indicated a bidirectional link between sleep and tics, which warrants consideration in clinical settings. Further research is needed to explore causal links.

Patients with Tourette syndrome frequently have sleep disturbances that may require polysomnographic testing. The use of deep brain stimulators is increasing with expanding indications including children with medically intractable Tourette syndrome. It is important to understand the effect the stimulator can have on polysomnographic monitoring. Herein we present an interesting case of an adolescent with medically intractable Tourette syndrome with a deep brain stimulator implant who underwent a polysomnogram demonstrating rhythmic, monomorphic artifact., Citation: Bindra T, Ingram DG. Images: Polysomnographic artifact in a patient with Tourette syndrome. J Clin Sleep Med . 2023;19(12):2149-2151., (© 2023 American Academy of Sleep Medicine.)

Comprehensive behavioral intervention for tics (CBIT) administered individually is an effective treatment for tics. However, the effectiveness of CBIT administered in groups for adults with Tourette syndrome and chronic tic disorders has not been investigated yet. This pilot study examined the effectiveness of group-based CBIT with respect to reduction of tic severity and tic-related impairment, as well as improvement of tic-related quality of life. Data from 26 patients were included in the intention-to-treat analyses. The Yale Global Tic Severity Scale was used to assess total tic severity and tic-related impairment. The Gilles de la Tourette - Quality of Life Scale was used to assess tic-related quality of life. These measures were administered at three points in time: at pretreatment, posttreatment, and 1-year follow-up. The results showed a significant reduction of total tic severity from pretreatment to 1-year follow-up, with larges effect sizes. Tic-related impairment and tic-related quality of life also improved significantly, although the effect sizes were smaller. Motor tics showed a stronger reduction than vocal tics. Additional analysis revealed that all change was achieved during treatment and that this effect was maintained from posttreatment to 1-year follow-up. The results of this study indicate that group-based CBIT is a promising treatment for tics., (© 2023 The Authors. Scandinavian Journal of Psychology published by Scandinavian Psychological Associations and John Wiley & Sons Ltd.)

Introduction: Premonitory urge (PU) is an aversive bodily sensation that signals the onset of tic disorder.To our knowledge, PU typically precedes the appearance of tic symptoms, and both age and tic severity are correlated with PU. However, inconsistent findings have also been reported. Hence, we conducted a meta-analysis to examine the relationship among premonitory symptoms, patient age and the severity of tic symptoms, as well as to summarize the research on the neural underpinnings of PU in Tourette syndrome (TS)., Methods: We conducted a literature search of relevant studies published between December 2005 and April 2022 using databases such as PubMed, Elsevier, PsycINFO, and Web of Science. Our analysis was carried out using R software with the assistance of the "meta" and "metafor" packages., Results: Our meta-analysis included 22 studies with a total of 1236 tic disorder patients. The mean Premonitory Urge for Tics Scale (PUTS) score was 20.17, with a 95% confidence interval of [18.14, 21.68]. Through meta-regression, we found that age and tic severity play important moderating roles in PU severity ( p < 0.0001). Neuroimaging studies suggest that PU is related to the insula, prefrontal cortex (PFC), anterior cingulate cortex (ACC), and supplementary motor area (SMA), regardless of the structural or functional level., Conclusions: Our meta-analysis confirmed the positive relationship between the severity of tics and PU and identified age as a significant factor influencing PU. The neural mechanisms underlying PU remain largely unknown, but evidence suggests that the insula, PFC, ACC, and SMA are related regions., Competing Interests: The authors declare no conflict of interest., (© 2023 The Author(s). Published by IMR Press.)

Background: Gills de la Tourette syndrome (TS) is a childhood-onset neurodevelopmental disorder manifested by motor and vocal tics. Kleefstra syndrome 1 (KS1), a rare genetic disorder, is caused by haploinsufficiency of the EHMT1 gene and is characterized by intellectual disability (ID), childhood hypotonia, and distinctive facial features. Tourette-like syndrome in KS1 has rarely been reported., Case Presentation: Here we describe a 7-year-old girl presenting involuntary motor and vocal tics, intellectual disability, childhood hypotonia, and dysmorphic craniofacial appearances, as well as comorbidities including attention deficit-hyperactivity disorder (ADHD), obsessive-compulsive disorder (OCD), and self-injurious behavior (SIB). The patient's CNV-seq testing revealed a de novo 320-kb deletion in the 9q34.3 region encompassing the EHMT1 gene., Conclusions: This is the first case reporting Tourette-like syndrome secondary to KS1 with a de novo microdeletion in the EHMT1 gene. Our case suggests TS with ID and facial anomalies indicate a genetic cause and broadens the phenotypic and genotypic spectrum of both TS and KS1., (© 2023. BioMed Central Ltd., part of Springer Nature.)

Aim: To compare transcranial magnetic stimulation (TMS)-derived measures of primary motor cortex (M1) physiology between children with and without Tourette syndrome, and to dimensionally analyze TMS measures with Tourette syndrome-related symptom severity., Method: We used a cross-sectional experimental design. Sixty 8- to 12-year-old children participated (30 with Tourette syndrome: three females, mean age 10 years 10 months, standard deviation [SD] 1 year 3 months; 30 typically developing children: seven females, mean age 10 years 7 months, SD 1 year 3 months). In the group with Tourette syndrome, 15 (one female, mean age 10 years 11 months, SD 1 year 3 months) had comorbid attention-deficit/hyperactivity disorder (ADHD), rated with the Conners, Third Edition and the parent-reported ADHD rating scales. Tic severity was rated with the Yale Global Tic Severity Scale and urge severity with the Individualized Premonitory Urge for Tics Scale. M1 short-interval cortical inhibition (SICI) and intracortical facilitation were compared between diagnostic groups and, within the group with Tourette syndrome, correlated with symptom severity using linear mixed-effects models for repeated measures., Results: Accounting for ADHD, we found no difference in SICI or intracortical facilitation in those with Tourette syndrome versus typically developing children (p > 0.1). In the group with Tourette syndrome, reduced M1 SICI predicted greater total (p = 0.012) and global (p = 0.002) tic severity. There were no associations with urge severity (p > 0.5)., Interpretation: Reduced M1 SICI is robustly associated with increased tic, but not urge, severity., What This Paper Adds: Increased tic severity is associated with reduced motor cortex short-interval cortical inhibition (SICI). Children with Tourette syndrome with increased urge severity also show increased tic severity. However, reduced motor cortex SICI is associated with tic, but not urge, severity., (© 2023 Mac Keith Press.)

Background: The microlesion effect refers to the improvement of clinical symptoms after deep brain stimulation (DBS) lead placement and is suggested to indicate optimal lead placement. Very few studies have reported its implications in neuropsychiatric disorders., Objective: To evaluate the magnitude of the microlesion effect in Tourette syndrome and the relationship between the microlesion effect and the anatomic location of implanted DBS leads., Methods: Six male patients were included. Their median age at surgery and follow-up period were 25 years (range, 18-47) and 12 months (range, 6-24), respectively. All patients were videotaped pre- and postoperatively, and tic frequencies were counted. We also analyzed the precision of lead placement and evaluated the normative connectome associated with the microlesion area., Results: The microlesion effect was observed as an improvement in tic symptoms in all patients, and the long-term clinical outcomes were favorable. The median motor tic frequency was 20.2 tics/min (range, 9.7-60) at baseline and decreased to 3.2 tics/min (1.2-11.3) in patients on postoperative day 1 ( P = .043) and to 5.7 tics/min (range, 1.9-16.6) in patients on postoperative day 7 ( P = .028). Phonic tic tended to improve immediately after surgery although the changes were not significant. Image analyses revealed that the precise position of the electrode was directed toward the anteromedial centromedian nucleus. Normative connectome analysis demonstrated connections between improvement-related areas and wide areas of the prefrontal cortex., Conclusion: This study shows that the microlesion effect may seem as an immediate improvement after optimal DBS lead placement in patients with Tourette syndrome., (Copyright © 2023 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the Congress of Neurological Surgeons.)

Objective: Among adults with Tourette syndrome, depression and anxiety symptoms are widely prevalent and consistently associated with poor quality of life. Important knowledge gaps remain regarding mood and anxiety dimensions of the adult Tourette syndrome phenotype. Taking a dimensional approach, this study sought to determine the prevalence, severity, and clinical correlates of depression and anxiety symptoms in a clinical sample of adults with Tourette syndrome and other chronic tic disorders., Methods: A retrospective chart review was conducted of all adults with a chronic tic disorder presenting to a tertiary care Tourette syndrome clinic between December 2020 and July 2022. Information extracted during chart review included data from scales administered as part of routine care: Quality of Life in Neurological Disorders (Neuro-QoL) Depression Short Form, Neuro-QoL Anxiety Short Form, Adult Attention-Deficit/Hyperactivity Disorder Self-Report Screening Scale, Dimensional Obsessive-Compulsive Scale, and Yale Global Tic Severity Scale. Relationships between variables were examined by conducting between-group, correlation, and multivariable regression analyses., Results: Data from 120 adult patients with a chronic tic disorder (77 men and 43 women) were analyzed. Neuro-QoL Anxiety scores were elevated in 66% of the cohort; Neuro-QoL Depression scores were elevated in 26%. Neuro-QoL Anxiety scores were significantly higher than general population norms, whereas Neuro-QoL Depression scores were not. After adjustment for covariates, depressive and anxiety symptom severity scores were significantly associated with each other and with obsessive-compulsive disorder symptom severity but not with tic severity. Sex-based differences emerged in the analyses., Conclusions: Among adults with chronic tic disorder, anxiety symptoms were more prevalent and severe than depressive symptoms, co-occurring psychiatric symptoms were more tightly linked with each other than with tic severity, and sex-based differences were evident., Competing Interests: Dr. Isaacs has received grant funding from Teva Branded Pharmaceutical Products, Research and Development. Dr. Riordan has received an educational grant from Medtronic. The other authors report no financial relationships with commercial interests.

Background and Purpose: Developmental stuttering and Tourette syndrome (TS) are common neurodevelopmental disorders. Although disfluencies may co-occur in TS, their type and frequency do not always represent pure stuttering. Conversely, core symptoms of stuttering may be accompanied by physical concomitants (PCs) that can be confused for tics. This scoping review aimed to explore the similarities and differences between stuttering and tics in terms of epidemiology, comorbidities, phenomenology, evolution, physiopathology, and treatment. We also described the nature of PCs in stuttering and disfluencies in TS., Methods: A literature search on Medline, Embase and PsycInfo was executed in March 2022. From 426 studies screened, 122 were included in the review (a majority being narrative reviews and case reports)., Results: TS and stuttering have several epidemiological, phenomenological, comorbidity, and management similarities suggesting shared risk factors and physiopathology (involving the basal ganglia and their connections with speech and motor control cortical regions). PCs in stuttering commonly involve the face (eyelids, jaw/mouth/lip movements) and sometimes the head, trunk and limbs. PCs can be present from early stages of stuttering and vary over time and within individuals. The function of PCs is unknown. Some individuals with TS have a distinct disfluency pattern, composed of a majority of typical disfluencies (mostly between-word disfluencies), and a mix of cluttering-like behaviors, complex phonic tics (e.g. speech-blocking tics, echolalia, palilalia), and rarely, atypical disfluencies., Conclusion: Future investigations are warranted to better understand the complex relationships between tics and stuttering and address the management of disfluencies in TS and PCs in stuttering., Competing Interests: Declaration of Competing Interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2023 Elsevier Inc. All rights reserved.)

Background and Purpose: The COVID-19 pandemic has been associated amongst other things with a sharp increase in adolescents and young adults presenting acutely with functional tics. Initial reports have suggested clinically relevant differences between functional tics and neurodevelopmental tics seen in primary tic disorders such as Tourette syndrome. We aimed to provide confirmatory findings from the largest single-centre cohort to date., Methods: In the present study we present data from 105 consecutive patients who developed functional tics during a 3-year period overlapping with the COVID-19 pandemic (April 2020-March 2023). All patients underwent a comprehensive neuropsychiatric assessment at a single specialist centre for tic disorders., Results: Female adolescents and young adults accounted for 69% of our sample. Functional tics had an acute/subacute onset in most cases (75% with a peak of severity within 1 month). We found a disproportionately high frequency of complex movements (81%) and vocalizations (75%). A subset of patients (23%) had a pre-existing primary tic disorder (Tourette syndrome with functional overlay). The most common psychiatric co-morbidities were anxiety (70%) and affective disorders (40%). Moreover, 41% of patients had at least one functional neurological disorder in addition to functional tics. Exposure to tic-related social media content was reported by half of the patients., Conclusions: Our findings confirm substantial clinical differences between functional tics developed during the pandemic and neurodevelopmental tics. Both patient- and tic-related red flags support the differential diagnostic process and inform ongoing monitoring in the post-pandemic era., (© 2023 The Authors. European Journal of Neurology published by John Wiley & Sons Ltd on behalf of European Academy of Neurology.)

Study Objectives: The aim of this mixed-methods study was to gain an insight through qualitative and quantitative means into the impact of the coronavirus disease 2019 (COVID-19) pandemic on children with and without Tourette syndrome (TS)., Methods: Parents/guardians of children and adolescents with TS (n = 95; mean age = 11.2 years, standard deviation = 2.68 years) and typically developing controls (n = 86; mean age = 10.7 years, standard deviation = 2.8 years) in the United Kingdom and Ireland completed an online questionnaire examining sleep, with open-ended questions pertaining to their perceived impact of COVID-19 on the sleep of their children. Nine items from the Sleep Disturbance Scale for Children were used to supplement qualitative data., Results: A negative impact of the pandemic on the sleep of both groups was observed, including exacerbated tics, sleep deprivation, and anxiety, with particular disruption for children with TS. Parents of children with TS reported poorer sleep patterns than parents of typically developing children on the Sleep Disturbance Scale for Children. Analyses showed that group and age predicted 43.8% of variance in sleep duration: F (4, 176) = 34.2, P < .001., Conclusions: Findings suggest that sleep patterns of children with TS may be more impacted by the pandemic than the average child. Given that there are generally more sleep issues reported in children with TS, further research is warranted in relation to the sleep health of children with TS in a postpandemic era. By identifying sleep issues potentially persisting after COVID-19, the true impact of the pandemic on the sleep of children and adolescents with Tourette syndrome may be ascertained., Citation: Colreavy E, Keenan L, Downes M. The impact of the COVID-19 pandemic on sleep in children with Tourette syndrome in Ireland and the United Kingdom. J Clin Sleep Med . 2023;19(8):1485-1493., (© 2023 American Academy of Sleep Medicine.)

Objective: Tourette syndrome (TS) is a neurobehavioral disorder characterized by motor and vocal tics. Simple tics are purposeless involuntary movements that spontaneously resolve during middle adolescence. Complex tics appear to be semi-voluntary movements that may become intractable when associated with obsessive-compulsive disorder (OCD). Sensory tics or urges preceded by tics suggest sensorimotor processing impairment in TS. We aimed to clarify its pathophysiology by exploring the pre-movement gating (attenuation) of somatosensory evoked potentials (SEPs)., Methods: We examined 42 patients (aged 9-48 years), 4 of whom underwent follow-up assessment, along with 19 healthy controls. We defined patients with only simple tics as TS-S and patients with complex tics as TS-C. Pre-movement gating of SEPs was assessed using a previously described method. Frontal N30 (FrN30) amplitudes were compared between pre-movement and resting states. The gating ratio of pre-movement/resting amplitude of the FrN30 component was assessed: the larger the ratio, the less the gating., Results: The gating ratio for TS-C patients was larger than that of TS-S patients and healthy controls, but a statistical difference between TS-S and TS-C appeared after 15 years and over (p < 0.001). There were no significant differences in the gating ratio between TS-S patients and healthy controls. The gating ratio was related to the severity of OCD (p < 0.05)., Conclusion: Sensorimotor processing was preserved for simple tics but impaired in complex tics, specifically after middle adolescence. Our study supports an age-dependent dysfunction of both motor and non-motor cortico-striato-thalamo-cortical circuits in complex tics. SEP gating seems promising as a tool for assessing age-dependent sensorimotor disintegration in TS., Competing Interests: Declaration of Competing Interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2023 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.)

Background: Tourette Syndrome (TS) is a childhood-onset neurodevelopmental disorder with a worldwide prevalence of about 0.3-1% of the population. During the pandemic caused by SARS-CoV-2 infection, the impact on the mental health of children and adolescents was very important. The persistence of symptoms in the post-acute phase of the disease has been termed Long COVID. The neuropsychiatric symptoms seem to be the most common impairment in children and adolescents with long COVID., Objectives: Considering the impact of pandemic on mental health, in this study we analyzed the long-term effects of SARS-CoV-2 infection in children and adolescents affected by TS., Methods: We conducted an online questionnaire covering socio-demographic and clinical data among 158 patients affected by TS or chronic tic disorders (CTD), of which 78 participants reported a positive SARS-CoV-2 infection. Data were collected to investigate tic severity and both the comorbidities, as well as lockdown-related changes to daily life activities and, in case of infection of SARS-CoV-2, possible symptoms of acute infection and long COVID. Markers of systemic inflammation including C-reactive protein (CRP), erythrocyte sedimentation rate (ESR), ferritin, iron, electrolytes, white blood cell counts, platelet cell counts levels, markers of liver, kidney and thyroid function were analyzed. First, all patients were screened with the Schedule for affective disorders and Schizophrenia for School age children-present and lifetime (Kiddie-SADS-PL) to rule out primary psychiatric disorders considered as criteria of exclusion. Then, all patients were clinically assessed at baseline (T0), and after three months (T1) through the administration of Yale Global Tic Severity Rating Scale (YGTSS), Multidimensional Anxiety Scale for Children (MASC), Child Depression Inventory (CDI) and Child Behavior Checklist (CBCL)., Results: Among the cohort of TS patients that contracted SARS-CoV-2 infection, 84.6% (n = 66) experienced any acute symptoms, and long COVID symptoms occurred in 38.5% (n = 30). A worsening of clinical symptoms of tics and eventually associated comorbidities occurred in 34.6% (n = 27) of TS patients that contracted SARS-CoV-2 infection. TS patients with or without SARS-CoV-2 infection showed an increase in the severity of tics and also behavioral, depressive and anxious symptoms. Instead, this increase was more evident in patients who contracted the infection than in patients who did not contract it., Conclusions: SARS-CoV-2 infection may have a role in the increase of tics and associated comorbidities in TS patients. Despite of these preliminary results, further investigations are necessary to improve knowledge about the acute and long-term impact of SARS-CoV-2 in TS patients., (© 2023. The Author(s).)

Background: Tourette syndrome (TS) is the most common cause of chronic tics. Patients with TS frequently manifest motor tics involving the eyes and face but oromandibular (OM) tics have been rarely studied., Materials and Methods: We reviewed the medical records and video-recordings of 155 consecutive patients with TS in our movement disorders clinic. In addition, we studied 35 patients with classic tardive dyskinesia (TD) and compared their clinical and demographic features with those with TS., Results: We identified 41 patients with OM tics (26.5%). Although patients with OM tics had a greater overall tic severity and higher frequency of.complex motor and phonic tics, in the bivariate analysis, only comorbid dystonic tics (P = 0.001), greater number of affected body parts (P = 0.012) and more frequent eye-rolling tics (P = 0.059) were included in the final regression model after controlling for other variables. When compared with patients with OM tics, patients with classic TD had more frequently masticatory movements (sensitivity, 0.86; specificity, 0.95), continuous tongue movements (sensitivity, 0.71; specificity, 1.0) and continuous OM movements (sensitivity, 0.4; specificity, 1.0)., Conclusions: OM tics are common and often troublesome or even disabling symptoms in patients with TS. They may be difficult to differentiate from TD, but the latter is typically manifested by continuous orolingual and masticatory movements., (© 2023. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany.)

Background: Sleep problems are common in children with Tourette Syndrome (TS). However, research regarding their demographic and clinical profile is limited., Methods: We examined characteristics of 114 children aged five to 17 years with a lifetime diagnosis of TS and compared children with sleep disorder (n = 32) and without sleep disorder (n = 82). Parent report from the 2014 National Survey of the Diagnosis and Treatment of ADHD and Tourette Syndrome provided demographics and clinical information, other diagnosed disorders, medication use, TS severity, and impairment., Results: More children with TS with sleep disorder were from households with lower parental education (P < 0.01) and poverty (P = 0.04); had other diagnoses (P = 0.03), including obsessive-compulsive disorder (P < 0.01), oppositional defiant disorder or conduct disorder (P < 0.01), attention-deficit/hyperactivity disorder (ADHD) (P = 0.02), and autism (P = 0.03); and had ever used TS medication (P = 0.01) than children with TS without sleep disorder. More children with TS with sleep disorder had severe TS symptoms (P <0.01), tic-related impairment (P<0.01), and severe ADHD symptoms (P < 0.01) compared with children with TS without sleep disorder., Conclusions: Findings suggest greater parent-reported impact and tic-related interference in children with TS with sleep disorder compared with TS without sleep disorder. Results underscore the importance of monitoring and intervention for TS exacerbations, other diagnosed disorders, and medication use, and consideration of socioeconomic context in sleep disorder management and prevention in children with TS., (Copyright © 2022 Elsevier Inc. All rights reserved.)

We aimed to compare tic- and non-tic-related impairment experienced by adolescent girls and boys (ages 13 through 17) with Tourette syndrome and associations with age. We extracted from the electronic health record child and parental responses to the mini-Child Tourette Syndrome Impairment Scale (mini-CTIM) and other questionnaire data reflective of tic- and non-tic-related impairment of adolescents with Tourette syndrome presenting to our clinic over a 12-month period. We identified a total of 132 (49 female, 83 male) unique adolescent encounters. Mini-CTIM scores did not differ significantly between genders. Tic- and non-tic-related impairment were lower in older boys, but not older girls. Obsessive-compulsive symptoms correlated with parent-reported non-tic-related impairment experienced by adolescent girls but not boys. During adolescence, tic- and non-tic-related impairments may be less likely to improve with age in girls. Future longitudinal studies are needed to confirm this finding.

Background: Previous studies have shown that Tourette syndrome (TS) has an impact on academic achievements. The aim of this study was to investigate the association between the severity of tics and comorbidities and educational outcomes., Methods: From 2005 to 2007, 395 participants were included in a large cohort (314 with TS and 81 controls) and the mean age was 12.60 ± 2.64 years. The cohort was re-examined after 4 to 8 years (median 5.6) where n  = 276 participants (223 with TS and 53 controls) were included with a mean age of 18.52 ± 2.73 years. At both time points, severity of tics and the presence and severity of psychiatric comorbidity were assessed. Educational achievements were assessed through structured interviews., Results: Children with TS had a lower passing rate at lower secondary and high school compared to healthy controls. More severe vocal tics were associated with fewer passing lower secondary school at a prospective level. At a cross-sectional level, more severe motor tics were associated with fewer passing high school. Tic severity only influenced children with TS without comorbidity. The severity of comorbidity was found to be associated with the educational level at a longitudinal view, but not cross-sectional., Conclusion: Overall, children with TS had a lower passing rate at lower secondary school and high school compared to healthy controls. We found that this difference was more likely driven by the severity of comorbidities than tic severity. It is important to be aware of academic achievement in children with TS in order to give them the right support and thereby optimize educational opportunities., Competing Interests: None declared., (Thieme. All rights reserved.)

Tourette syndrome and chronic tic disorders are characterized by the presence of tics. Different behavioral therapies have shown to be efficacious for treating tics in children and adolescents, but Exposure and Response Prevention (ERP) is a less researched method. However, ERP is a method often used in the clinical setting. Therefore, the present study evaluated the severity of tics over time from beginning of ERP to follow-up approximately 1 year after last training session.In total, 116 patients treated with ERP face to face or ERP via web-based videoconferencing were included. The primary outcome measure was tic severity measured with the Danish version of the Yale Global Tic Severity Scale.The results showed that tic severity decreased during ERP and lasted in the follow-up period, with a statistically higher decrease in the group with patients who completed ERP as planned and the group that stopped earlier than planned because of reduction in tics, compared with those who dropped out due to lack of motivation ( p  < 0.001).The study concludes that ERP seems to have an immediate and a long-term effect on severity of tics, especially in those who complete the program or those who discontinue earlier due to good results., Competing Interests: None declared., (Thieme. All rights reserved.)

Tourette syndrome (TS) is a childhood-onset disorder in which tics are often preceded by premonitory sensory urges. More severe urges correlate with worse tics and can render behavioral therapies less effective. The supplementary motor area (SMA) is a prefrontal region believed to influence tic performance. To determine whether cortical physiological properties correlate with urges and tics, we evaluated, in 8-12-year-old right-handed TS children (n = 17), correlations of urge and tic severity scores and compared both to cortical excitability (CE) and short- and long-interval cortical inhibition (SICI and LICI) in both left and right M1. We also modeled these M1 transcranial magnetic stimulation measures with SMA gamma-amino butyric acid (GABA) levels in TS and typically developing control children (n = 16). Urge intensity correlated strongly with tic scores. More severe urges correlated with lower CE and less LICI in both right and left M1. Unexpectedly, in right M1, lower CE and less LICI correlated with less severe tics. We found that SMA GABA modulation of right, but not left, M1 CE and LICI differed in TS. We conclude that in young children with TS, lower right M1 CE and LICI, modulated by SMA GABA, may reflect compensatory mechanisms to diminish tics in response to premonitory urges., (© The Author(s) 2022. Published by Oxford University Press. All rights reserved. For permissions, please e-mail: journals.permission@oup.com.)

Background: Although Deep Brain Stimulation (DBS) is a safe and proven treatment modality for patients suffering from debilitating movement and neuropsychiatric disorders, it is not free from complications. Management of skin erosion and infection following DBS surgery constitutes a challenge in everyday clinical practice., Objectives: Skin-related complications were evaluated in patients who underwent DBS surgery due to Parkinson's disease (PD), dystonia, essential tremor (ET), and other indications including Tourette syndrome (TS), Obsessive-Compulsive Disorder (OCD), and epilepsy., Methods: A retrospective analysis of clinical data was performed on patients who underwent DBS surgery between November 2008 and September 2021 at the Department of Neurosurgery, Institute of Psychiatry and Neurology, Warsaw., Results: 525 patients who underwent 927 DBS leads implantations were included in the analysis. There were 398 patients with PD, 80 with dystonia, 26 with ET, 7 with drug-resistant epilepsy, 5 with Multiple Sclerosis, 4 with Holme's or cerebellar tremor, 3 with TS, and 2 with OCD. 42 patients (8,0%) had 78 skin infection episodes. The overall level of skin erosion was 3,8% (20/525 patients). The risk of developing infection episode was connected with younger age at diagnosis (p = 0.017) and at surgery (p = 0.023), whereas the development of skin erosion was connected with the dystonia diagnosis (p = 0.012). Patients with dystonia showed the highest rate of infections and erosions (11/70 and 7/70 patients retrospectively)., Discussion: Postoperative skin complications are a serious side effect of DBS surgery., Conclusion: Our study suggests that dystonic patients are at higher risk of developing skin-related complications after DBS surgery., Competing Interests: Conflict of interest The authors declare no potential conflict of interest., (Copyright © 2023 Elsevier B.V. All rights reserved.)

Background and Objectives: All US Food and Drug Administration-approved medications for Tourette syndrome are antipsychotics, and their use is limited by the risk of weight gain, metabolic changes, and drug-induced movement disorders. Several small trials suggest that ecopipam, a first-in-class, selective dopamine 1 receptor antagonist, reduces tics with a low risk for these adverse events. This trial sought to further evaluate the efficacy, safety, and tolerability of ecopipam in children and adolescents with moderate to severe Tourette syndrome., Methods: This was a multicenter, randomized, double-blind, placebo-controlled, phase 2b trial. Subjects aged ≥6 to <18 years with a baseline Yale Global Tic Severity Score Total Tic Score of ≥20 were randomly assigned 1:1 to ecopipam (n = 76) or placebo (n = 77). The primary endpoint was mean change over 12 weeks in the Yale Global Tic Severity Score Total Tic Score. The Clinical Global Impression of Tourette Syndrome Severity was the secondary endpoint. Safety and tolerability were evaluated at each study visit., Results: Total tic scores were significantly reduced from baseline to 12 weeks in the ecopipam group compared with placebo (least squares mean differences -3.44, 95% confidence interval -6.09 to -0.79, P = .01). Improvement in Clinical Global Impression of Tourette Syndrome Severity was also greater in the ecopipam group (P = .03). More weight gain was seen in subjects assigned to placebo. No metabolic or electrocardiogram changes were identified. Headache (15.8%), insomnia (14.5%), fatigue (7.9%), and somnolence (7.9%) were the most common adverse events., Conclusions: Among children and adolescents with TS, ecopipam reduces tics to a greater extent than placebo, without observable evidence of common antipsychotic-associated side effects.

The following case report outlines the impact of motor tics linked to Tourette's syndrome on dental development and healing following a dental injury to a maxillary central incisor. Emergency care and splinting of a mobile extruded maxillary left central incisor tooth was carried out at the local dentist on the same day as the dental trauma. A subsequent referral was made to the paediatric dental department for continued mobility of the maxillary central incisor on splint removal approximately 2 weeks later. A clinical and radiographic examination revealed shortened root length and apical root blunting associated with both maxillary anterior teeth. Further questioning revealed the likely cause of this to be related to the clenching and biting oral tics which the patient has experienced over the past 4 years. A removable splint has been fabricated for night-time wear and a mouthgaurd has been recommended for use during contact sports. Regular reviews will be conducted using a shared care approach between the patient's local dentist and the paediatric dental department., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2023. No commercial re-use. See rights and permissions. Published by BMJ.)

In the last years, much focus has been given to the possible role of inflammatory and immunologic alterations in the pathophysiology of obsessive-compulsive disorder (OCD) and some related conditions, such as pediatric autoimmune neuropsychiatric disorders associated with streptococcal infection (PANDAS) and Tourette syndrome (TS). Although the matter is intriguing, the available data are still controversial and/or limited. Therefore, the aim of this chapter was at reviewing and commenting on the literature on possible dysfunctions of inflammatory and immune system processes in OCD, PANDAS, and TS.This narrative review was carried out through searching PubMed and Google Scholar for English language papers from January 1985 to December 31, 2021.The data gathered up to now would suggest that the mechanisms involved might be heterogeneous according to the age of the patients and the disorder examined. Indeed, PANDAS seem more related to infections triggering autoimmunity not necessarily following group A beta-hemolytic streptococcal (GABHS) infection, as supposed in the past. Autoimmunity seems also important in TS, if coupled with an individual vulnerability that can be genetic and/or environmental. The data in adult OCD, albeit scattered and sometimes obtained in small samples of patients, would indicate that immune system and inflammatory processes are involved in the pathophysiology of the disorder. However, it is still unclear to conclude whether they are primary or secondary phenomena.In conclusion, taken together, the current findings pave that way towards novel and promising domains to explore the pathophysiology of OCD and related disorders, as well towards the development of innovative therapeutic strategy beyond current pharmacological paradigms., (© 2023. The Author(s), under exclusive license to Springer Nature Singapore Pte Ltd.)

Tics peak in late childhood and decline during adolescence. Yet, for some with Tourette's disorder, tics persist into adulthood. We evaluated childhood predictors of adult tic severity and tic impairment, and change over time. Eighty adolescents/adults were evaluated 11 years following a randomized-controlled trial of behavior therapy. An independent evaluator rated tic severity and tic impairment at baseline, posttreatment, and long-term follow-up. At baseline, parents completed demographics/medical history, and youth tic, internalizing, and externalizing symptom ratings. Youth rated premonitory urge severity and family functioning. After controlling for prior tic treatment effects, female sex and higher tic severity predicted higher tic severity in adulthood; and female sex, no stimulant medication use, higher tic severity, and poorer family functioning predicted higher tic impairment. Higher tic severity and premonitory urge severity predicted smaller reductions in tic severity, whereas higher externalizing symptoms predicted greater reduction in tic severity. Female sex predicted smaller reduction in tic impairment, and externalizing symptoms predicted greater reduction in tic impairment. Female sex and childhood tic severity are important predictors of tic severity and tic impairment in adulthood. Family functioning, premonitory urge severity, and tic severity are important modifiable targets for early or targeted intervention to improve long-term outcomes., (Copyright © 2022. Published by Elsevier Ltd.)

Background: Dystonic tics differ from clonic tics by their slower and more sustained nature. Dystonic tics are often present in patients with Tourette syndrome (TS) and other tic-disorders. However, their phenomenology and impact on overall impairment have not been extensively studied., Materials and Methods: We assessed clinical history and tic duration in video-recordings from patients with TS evaluated at our movement disorders clinic. Dystonic tics were defined as those lasting ≥ 1000 ms (ms)., Results: Of the total of 201 patients with TS, there were 156 with video-recordings suitable for tic duration analysis, of their tics, 57 (36.5%) of whom had dystonic motor tics, including 9 (5.7%) with dystonic phonic tics. Dystonic motor tics had a duration range between 1033 and 15,000 ms and dystonic phonic tics between 1132 and 17,766 ms. Patients with dystonic tics were older 24.4 vs. 16.5 years (P = 0.005) and had an older age at onset 12.9 vs. 7.2 years (P < 0.001), than patients without dystonic tics. The bivariate analysis showed an association between the presence of dystonic tics, greater tic severity and wider body distribution. The multivariate regression analysis showed a statistical association with older age at evaluation (P = 0.001), greater tic severity on video-recordings (P = 0.001) and co-occurrence with complex motor tics (P = 0.020). The presence of dystonic tics increased the risk for being considered for deep brain stimulation therapy, odds ratio: 15.7 (P = 0.002)., Conclusion: Dystonic tics, observed in about a third of patients with TS, are associated with increased severity of TS., (© 2022. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany.)

Premonitory urges preceding tics are a cardinal feature of Gilles de la Tourette syndrome (GTS), a developmental disorder usually starting during middle childhood. However, the temporal relation between urges and tics has only been investigated in adults. In 25 children and adolescents with GTS (8-18 years), we assess urge-tic associations, including inter-individual differences, correlation to clinical measures, and in comparison to a previously reported sample of adult GTS patients. Group-level analyses confirmed positive associations between urges and tics. However, at the individual level, less than half of participants showed positive associations, a similar proportion did not, and in two participants, the association was reversed. Tic expression and subjective urge levels correlated with corresponding clinical scores and participants with more severe tics during the urge monitor exhibited stronger urge-tic associations. Associations between reported urge levels and instantaneous tic intensity tended to be less pronounced in children and adolescents than in adult GTS patients. The observed heterogeneity of urge-tic associations cast doubt on the notion that tics are directly caused by urges. More severe tics may facilitate anticipation of tics and thereby lead to more pronounced urge-tic associations, consistent with a hypothesis of urges as a byproduct of tics., (© 2022. The Author(s).)

Objective: To evaluate the prevalence of nail biting in child and adolescent outpatients at a single institution and the chronological relationship between nail biting and tics in patients with Tourette syndrome (TS) with or without attention-deficit hyperactivity disorder (ADHD)., Design: Retrospective observational study., Setting: Teaching hospital in Taiwan., Participants: All participants were aged 4-18 years, including 535 patients with TS, 230 patients with provisional tic disorder and 1460 patients without neurological or psychiatric disorders (controls)., Outcome Measures: Presence of nail biting, starting age for nail biting and starting age for motor and/or vocal tics., Results: Nail biting was more commonly observed in patients with TS (56.6%) than in patients with provisional tic disorder (27.4%) or controls (15.0%), regardless of sex (all p<0.020). Nail biting was also more common in patients with TS with ADHD than in those without (75.0% vs 47.6%; p<0.001), but the starting age was significantly later in those with concomitant ADHD than without (5.3 vs 3.8 years; p<0.001). In patients with TS, the onset of nail biting occurred earlier than that of tics, regardless of ADHD status., Conclusion: Nail biting was more prevalent and occurred earlier than tics in patients with TS, regardless of ADHD status, in the study population., Competing Interests: Competing interests: None declared., (© Author(s) (or their employer(s)) 2022. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.)

Tourette's syndrome affects about 1% of the school-age population. Children and young people with the condition exhibit involuntary motor and vocal (also known as phonic) tics. Only 10% of those with Tourette's syndrome have coprolalia, which involves involuntary swearing and inappropriate language. Tourette's syndrome often presents with other conditions such as obsessive-compulsive disorder, autism and attention deficit hyperactivity disorder. Those affected by Tourette's syndrome may be able to manage the condition with little or no professional support, but some may need pharmacological and nonpharmacological input, particularly from mental health services. It is essential that peers, teachers, healthcare staff and members of the public are aware of Tourette's syndrome to ensure that children and young people living with the condition are supported well., Competing Interests: None declared, (© 2022 RCN Publishing Company Ltd. All rights reserved. Not to be copied, transmitted or recorded in any way, in whole or part, without prior permission of the publishers.)

Background: Oculogyric tics are identified in a substantial proportion of patients with Tourette syndrome (TS) and related tic-disorders. Nevertheless, studies assessing its frequency and clinical correlations are lacking., Materials and Methods: We reviewed video-recordings and clinical history of 201 patients with TS and chronic motor/phonic tic disorder, diagnosed according to DSM-5., Results: Oculogyric tics presented in 22.4% of our patients. Transient upward gaze was the most common phenomenology in patients with simple oculogyric tics; whereas eye-closure followed by upward and lateral deviations was the most commonly observed phenomenon in patients with combined oculogyric tics. Oculogyric tics have a median duration of 799 ms (range 299-34,500 ms). Patients with oculogyric tics were younger (P = 0.023) and had a higher frequency of cranial tics (P = 0.037) compared to those without oculogyric tics. No differences in tic severity, frequency of attention-deficit/hyperactivity disorder, obsessive-compulsive disorder or use of dopamine receptor antagonists were observed in patients with and without oculogyric tics., Conclusions: Oculogyric tics are a common phenomenology in chronic tic disorders. They are more common in younger patients with TS and are markers of other cranial tics, but not of TS severity or comorbid neuropsychiatric syndromes., (© 2022. The Author(s) under exclusive licence to Belgian Neurological Society.)

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., Competing Interests: The authors declare no conflict of interest., (© 2022 The Author(s). Published by IMR Press.)

Introduction: Tourette syndrome (TS) is characterized by the presence of motor and phonic tics, as well as a variety of behavioral co-morbidities. Self-injurious behavior (SIB) is one of the most serious manifestations of TS, but its pathophysiology is poorly understood., Methods: Consecutive patients with TS studied in a tertiary care center., Results: We identified a total of 34 patients (16.9%) with SIB from a cohort of 201 patients with TS. Most of these patients (n = 23, 11.4%) experienced self-directed damage; while others had outward-directed (n = 7, 3.5%) or tic-related SIB (n = 4, 2%). Compared to other patients with TS, those who manifested SIB (self- and outward-directed damage) were more likely to have tics involving shoulder (P = 0.046), trunk (P = 0.006), and arm (P = 0.017); as well as dystonic tics (P = 0.016); complex motor tics (P < 0.001), copropraxia (P = 0.045), complex phonic tics (P = 0.003), higher number of phonic tics (P = 0.001), verbalizations (P = 0.001), coprolalia (P = 0.006) and obsessive compulsive disorder (OCD) (P < 0.001) as determined by bivariate analysis. In the multivariate analysis only complex motor tics (P = 0.006), obsessive-compulsive behavior (P = 0.025) and greater severity of tics (P = 0.002) showed a statistically significant association with SIB. Patients with SIB had a greater probability of being selected for deep brain stimulation (DBS) therapy by the treating clinician (P = 0.01)., Conclusions: SIB is observed in about 17% of patients with TS. The presence of complex motor tics, OCD and greater severity of tics was related to the presence of SIB., (© 2021. Springer-Verlag GmbH Germany, part of Springer Nature.)

Introduction: Premonitory urges in Tourette disorder are often linked to altered somatosensory processing, which might include deficits in metacognition. We explored tactile and visual metacognitive ability in people with Tourette disorder and healthy control participants., Methods: Patients with Tourrete disorder and healthy control participants completed a tactile and a visual metacognitive task. On each trial, participants did a forced choice discrimination and then rated their confidence in their decision. To quantify metacognitive ability, we used m-ratio - a bias-free measure that allows for comparisons across modalities. Correlations between severity of tics and premonitory urges with tactile metacognitive sensitivity were also performed., Results: Metacognitive ability in both tactile and visual domains was comparable between adults with Tourette disorder and healthy controls. We also found no evidence for correlations between tactile metacognitive ability and severity of premonitory urges or tic severity., Conclusions: Tactile and visual metacognition is not impaired in adults with Tourette disorder. These results question the role of altered tactile metacognition in pathophysiology of tic disorders., (Copyright © 2022 Elsevier Ltd. All rights reserved.)