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1.
No Shinkei Geka ; 52(3): 619-629, 2024 May.
Article in Japanese | MEDLINE | ID: mdl-38783505

ABSTRACT

The efficacy of deep brain stimulation(DBS)for Tourette's syndrome is being well established. Herein, we performed DBS in 38 patients and confirmed that its efficacy was comparable with that reported internationally. Although many patients experience severe symptoms, the indications for surgery remain controversial. One reason for this is that Tourette syndrome has the potential for spontaneous remission, while DBS treatment results in the need for long-term management, which can be difficult for some patients. Furthermore, while several targets for DBS have been reported, no treatment guidelines have yet been established. The efficacy of DBS for neuropsychiatric disorders, such as obsessive-compulsive disorder, depression, and dementia, is gradually being reported. However, this use has many limitations in terms of expectations similar to those seen with Tourette's syndrome, leading to problems with expanding indications for these disorders. Indications for these disorders should be addressed in conjunction with ethical issues. It is expected that more data on this topic will be collected in the future.


Subject(s)
Deep Brain Stimulation , Tourette Syndrome , Humans , Tourette Syndrome/therapy , Adult , Male , Female , Mental Disorders/therapy , Obsessive-Compulsive Disorder/therapy , Middle Aged , Treatment Outcome , Adolescent , Aged
2.
JAMA Netw Open ; 7(5): e248468, 2024 May 01.
Article in English | MEDLINE | ID: mdl-38700867

ABSTRACT

Importance: Behavior therapy is a recommended intervention for Tourette syndrome (TS) and chronic tic disorder (CTD), but availability is limited and long-term effects are uncertain. Objective: To investigate the long-term efficacy and cost-effectiveness of therapist-supported, internet-delivered exposure and response prevention (ERP) vs psychoeducation for youths with TS or CTD. Design, Setting, And Participants: This 12-month controlled follow-up of a parallel group, superiority randomized clinical trial was conducted at a research clinic in Stockholm, Sweden, with nationwide recruitment. In total, 221 participants aged 9 to 17 years with TS or CTD were enrolled between April 26, 2019, and April 9, 2021, of whom 208 (94%) provided 12-month follow-up data. Final follow-up data were collected on June 29, 2022. Outcome assessors were masked to treatment allocation throughout the study. Interventions: A total of 111 participants were originally randomly allocated to 10 weeks of therapist-supported, internet-delivered ERP and 110 participants to therapist-supported, internet-delivered psychoeducation. Main Outcomes And Measures: The primary outcome was within-group change in tic severity, measured by the Total Tic Severity Score of the Yale Global Tic Severity Scale (YGTSS-TTSS), from the 3-month follow-up to the 12-month follow-up. Treatment response was defined as 1 (very much improved) or 2 (much improved) on the Clinical Global Impression-Improvement scale. Analyses were intention-to-treat and followed the plan prespecified in the published study protocol. A health economic evaluation was performed from 3 perspectives: health care organization (including direct costs for treatment provided in the study), health care sector (additionally including health care resource use outside of the study), and societal (additionally including costs beyond health care [eg, parent's absenteeism from work]). Results: In total, 221 participants were recruited (mean [SD] age, 12.1 [2.3] years; 152 [69%] male). According to the YGTSS-TTSS, there were no statistically significant changes in tic severity from the 3-month to the 12-month follow-up in either group (ERP coefficient, -0.52 [95% CI, -1.26 to 0.21]; P = .16; psychoeducation coefficient, 0.00 [95% CI, -0.78 to 0.78]; P > .99). A secondary analysis including all assessment points (baseline to 12-month follow-up) showed no statistically significant between-group difference in tic severity from baseline to the 12-month follow-up (coefficient, -0.38 [95% CI, -1.11 to 0.35]; P = .30). Treatment response rates were similar in both groups (55% in ERP and 50% in psychoeducation; odds ratio, 1.25 [95% CI, 0.73-2.16]; P = .42) at the 12-month follow-up. The health economic evaluation showed that, from a health care sector perspective, ERP produced more quality-adjusted life years (0.01 [95% CI, -0.01 to 0.03]) and lower costs (adjusted mean difference -$84.48 [95% CI, -$440.20 to $977.60]) than psychoeducation at the 12-month follow-up. From the health care organization and societal perspectives, ERP produced more quality-adjusted life years at higher costs, with 65% to 78% probability of ERP being cost-effective compared with psychoeducation when using a willingness-to-pay threshold of US $79 000. Conclusions And Relevance: There were no statistically significant changes in tic severity from the 3-month through to the 12-month follow-up in either group. The ERP intervention was not superior to psychoeducation at any time point. While ERP was not superior to psychoeducation alone in reducing tic severity at the end of the follow-up period, ERP is recommended for clinical implementation due to its likely cost-effectiveness and support from previous literature. Trial Registration: ClinicalTrials.gov Identifier: NCT03916055.


Subject(s)
Cost-Benefit Analysis , Tourette Syndrome , Humans , Tourette Syndrome/therapy , Male , Female , Child , Adolescent , Follow-Up Studies , Internet , Sweden , Treatment Outcome , Internet-Based Intervention , Behavior Therapy/methods , Behavior Therapy/economics
3.
BMC Med ; 22(1): 218, 2024 May 30.
Article in English | MEDLINE | ID: mdl-38816877

ABSTRACT

BACKGROUND: Deep brain stimulation (DBS) is a promising therapy for refractory Gilles de la Tourette syndrome (GTS). However, its long-term efficacy, safety, and recommended surgical age remain controversial, requiring evidence to compare different age categories. METHODS: This retrospective cohort study recruited 102 GTS patients who underwent DBS between October 2006 and April 2022 at two national centers. Patients were divided into two age categories: children (aged < 18 years; n = 34) and adults (aged ≥ 18 years; n = 68). The longitudinal outcomes as tic symptoms were assessed by the YGTSS, and the YBOCS, BDI, and GTS-QOL were evaluated for symptoms of obsessive-compulsive disorder (OCD), depression, and quality of life, respectively. RESULTS: Overall, these included patients who finished a median 60-month follow-up, with no significant difference between children and adults (p = 0.44). Overall, the YGTSS total score showed significant postoperative improvements and further improved with time (improved 45.2%, 51.6%, 55.5%, 55.6%, 57.8%, 61.4% after 6, 12, 24, 36, 48, and ≥ 60 months of follow-up compared to baseline, respectively) in all included patients (all p < 0.05). A significantly higher improvement was revealed in children than adults at ≥ 60 months of follow-up in the YGTSS scores (70.1% vs 55.9%, p = 0.043), and the time to achieve 60% improvement was significantly shorter in the children group (median 6 months vs 12 months, p = 0.013). At the last follow-up, the mean improvements were 45.4%, 48.9%, and 55.9% and 40.3%, 45.4%, and 47.9% in YBOCS, BDI, and GTS-QOL scores for children and adults, respectively, which all significantly improved compared to baseline (all p < 0.05) but without significant differences between these two groups (all p > 0.05), and the children group received significantly higher improvement in GTS-QOL scores than adults (55.9% vs. 47.9%, p = 0.049). CONCLUSIONS: DBS showed acceptable long-term efficacy and safety for both children and adults with GTS. Surgeries performed for patients younger than 18 years seemed to show acceptable long-term efficacy and safety and were not associated with increased risks of loss of benefit compared to patients older than 18 at the time of surgery. However, surgeries for children should also be performed cautiously to ensure their refractoriness and safety.


Subject(s)
Deep Brain Stimulation , Tourette Syndrome , Humans , Tourette Syndrome/therapy , Deep Brain Stimulation/methods , Male , Female , Child , Adult , Adolescent , Retrospective Studies , Follow-Up Studies , Young Adult , Treatment Outcome , Quality of Life , Middle Aged , Age Factors
4.
Curr Opin Pediatr ; 36(3): 331-341, 2024 06 01.
Article in English | MEDLINE | ID: mdl-38655812

ABSTRACT

PURPOSE OF REVIEW: We highlight novel and emerging therapies in the treatment of childhood-onset movement disorders. We structured this review by therapeutic entity (small molecule drugs, RNA-targeted therapeutics, gene replacement therapy, and neuromodulation), recognizing that there are two main approaches to treatment: symptomatic (based on phenomenology) and molecular mechanism-based therapy or 'precision medicine' (which is disease-modifying). RECENT FINDINGS: We highlight reports of new small molecule drugs for Tourette syndrome, Friedreich's ataxia and Rett syndrome. We also discuss developments in gene therapy for aromatic l-amino acid decarboxylase deficiency and hereditary spastic paraplegia, as well as current work exploring optimization of deep brain stimulation and lesioning with focused ultrasound. SUMMARY: Childhood-onset movement disorders have traditionally been treated symptomatically based on phenomenology, but focus has recently shifted toward targeted molecular mechanism-based therapeutics. The development of precision therapies is driven by increasing capabilities for genetic testing and a better delineation of the underlying disease mechanisms. We highlight novel and exciting approaches to the treatment of genetic childhood-onset movement disorders while also discussing general challenges in therapy development for rare diseases. We provide a framework for molecular mechanism-based treatment approaches, a summary of specific treatments for various movement disorders, and a clinical trial readiness framework.


Subject(s)
Movement Disorders , Child , Humans , Deep Brain Stimulation , Friedreich Ataxia/therapy , Friedreich Ataxia/genetics , Genetic Therapy/methods , Movement Disorders/therapy , Precision Medicine/methods , Rett Syndrome/genetics , Rett Syndrome/therapy , Tourette Syndrome/therapy , Tourette Syndrome/genetics
5.
J Psychiatr Res ; 173: 34-40, 2024 May.
Article in English | MEDLINE | ID: mdl-38479346

ABSTRACT

There is growing enthusiasm for the treatment of neuropsychiatric disorders using neuromodulation. While some reports claim that transcranial magnetic stimulation (TMS) can be used to treat Tourette Syndrome (TS), little research exists to support this assertion. This meta-analysis examined the efficacy of TMS to reduce tic severity in patients with TS. Additionally, it explored the effect of TMS to reduce premonitory urge severity-the primary mechanism implicated in the frontline evidence-based treatment of TS. Five treatment comparisons were selected using PRISMA guidelines. All studies included were required to be (1) a randomized controlled trial, (2) compare TMS to a sham condition, and (3) have all participants meet diagnostic criteria for a persistent tic disorder and/or TS. A random effects model meta-analysis examined the efficacy of using TMS to reduce tic severity and explored the effect of TMS to reduce premonitory urge severity. TMS did not significantly reduce tic severity (g = 0.44; 95% CI = -0.17, 1.05; z = 1.40; p = 0.16), but a moderate reduction in premonitory urge severity was found (g = 0.63; 95% CI = 0.9, 1.17; z = 2.27; p < 0.02). Trials with larger sample sizes and a preponderance of women were found to have greater therapeutic effects of TMS for tic severity. There is limited support for the use of TMS to reduce tic severity, though reductions in premonitory urge severity were observed. Major limitations of the existing literature are examined, with a call for research investigating newer TMS protocols and their use as a treatment augmentation strategy.


Subject(s)
Tic Disorders , Tics , Tourette Syndrome , Female , Humans , Emotions , Randomized Controlled Trials as Topic , Tic Disorders/therapy , Tourette Syndrome/therapy , Transcranial Magnetic Stimulation , Male
6.
Sci Rep ; 14(1): 6467, 2024 03 18.
Article in English | MEDLINE | ID: mdl-38499664

ABSTRACT

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.


Subject(s)
Deep Brain Stimulation , Tics , Tourette Syndrome , Humans , Male , Female , Tourette Syndrome/therapy , Tourette Syndrome/complications , Tics/therapy , Tics/etiology , Deep Brain Stimulation/methods , Treatment Outcome , Severity of Illness Index
7.
Neurosci Biobehav Rev ; 159: 105609, 2024 Apr.
Article in English | MEDLINE | ID: mdl-38447821

ABSTRACT

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.


Subject(s)
Tic Disorders , Tics , Tourette Syndrome , Humans , Tics/etiology , Tics/therapy , Tourette Syndrome/complications , Tourette Syndrome/diagnosis , Tourette Syndrome/therapy , Emergencies , Tic Disorders/epidemiology , Comorbidity
8.
Zhongguo Zhen Jiu ; 44(3): 343-350, 2024 Mar 12.
Article in English, Chinese | MEDLINE | ID: mdl-38467512

ABSTRACT

OBJECTIVES: To explore the rules of acupoint selection and compatibility of acupuncture for Tourette syndrome(TS) in children. METHODS: The relevant literature regarding acupuncture for Tourette syndrome in children included in CNKI, Wanfang, VIP, SinoMed, PubMed, Web of Science and Cochrane Library from the establishment of the database to March 31st, 2023 was retrieved.The information of acupuncture prescription, syndrome type, meridian affinity was extracted to set up database. The Microsoft Excel 2019 was used for descriptive statistical analysis, SPSS modeler18.0 was for association rule analysis, lantern5.0 was for latent structure analysis and comprehensive clustering. RESULTS: ①A total of 80 literature was included, and 112 acupuncture prescriptions were extracted, involving 104 acupoints, with a cumulative frequency of 859 times.②The acupoints with high use frequency were Taichong(LR 3), Baihui(GV 20), Fengchi(GB 20), Hegu(LI 4), Sishencong(EX-HN 1), Sanyinjiao(SP 6) and Zusanli(ST 36).③In the treatment of TS with acupuncture, the governor vessel acupoints were the most frequently used, the proportion of acupoints on the head, face, neck and lower limbs was higher. ④The association rule analysis showed that Fengchi(GB 20)-Hegu(LI 4) and Taichong(LR 3)-Hegu(LI 4) had the highest support degree, both were 47.32%.⑤Five comprehensive clustering models were obtained by analyzing the latent structure of high-frequency acupoints, corresponding to yin deficiency disturbing wind, liver hyperactivity and spleen deficiency, liver yang transforming into wind, phlegm-heat harassing the interior and qi stagnation transformed fire. CONCLUSIONS: Acupuncture for TS in children is based on the principle of soothe the liver and extinguish the wind, regulating qi and blood, and paying attention to regulating spirit and qi. The core acupoints are Fengchi(GB 20), Hegu(LI 4), Taichong(LR 3), Baihui(GV 20), Sanyinjiao(SP 6) , Zusanli(ST 36), acupoints should be selected according to different syndrome in clinical.


Subject(s)
Acupuncture Therapy , Meridians , Tourette Syndrome , Child , Humans , Acupuncture Points , Tourette Syndrome/therapy , Databases, Factual
10.
Brain Stimul ; 17(2): 197-201, 2024.
Article in English | MEDLINE | ID: mdl-38341176

ABSTRACT

BACKGROUND: Deep brain stimulation (DBS) of the thalamus can effectively reduce tics in severely affected patients with Tourette syndrome (TS). Its effect on cortical oscillatory activity is currently unknown. OBJECTIVE: We assessed whether DBS modulates beta activity at fronto-central electrodes. We explored concurrent EEG sources and probabilistic stimulation maps. METHODS: Resting state EEG of TS patients treated with thalamic DBS was recorded in repeated DBS-on and DBS-off states. A mixed linear model was employed for statistical evaluation. EEG sources were estimated with eLORETA. Thalamic probabilistic stimulation maps were obtained by assigning beta power difference scores (DBS-on minus DBS-off) to stimulation sites. RESULTS: We observed increased beta power in DBS-on compared to DBS-off states. Modulation of cortical beta activity was localized to the midcingulate cortex. Beta modulation was more pronounced when stimulating the thalamus posteriorly, peaking in the ventral posterior nucleus. CONCLUSION: Thalamic DBS in TS patients modulates beta frequency oscillations presumably important for sensorimotor function and relevant to TS pathophysiology.


Subject(s)
Beta Rhythm , Deep Brain Stimulation , Thalamus , Tourette Syndrome , Humans , Tourette Syndrome/therapy , Tourette Syndrome/physiopathology , Deep Brain Stimulation/methods , Male , Thalamus/physiopathology , Thalamus/physiology , Adult , Beta Rhythm/physiology , Female , Electroencephalography , Young Adult , Cerebral Cortex/physiopathology , Cerebral Cortex/physiology , Middle Aged , Adolescent
11.
Behav Ther ; 55(2): 263-276, 2024 Mar.
Article in English | MEDLINE | ID: mdl-38418039

ABSTRACT

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


Subject(s)
Tic Disorders , Tics , Tourette Syndrome , Humans , Tics/therapy , Tic Disorders/psychology , Tourette Syndrome/therapy , Tourette Syndrome/psychology , Behavior Therapy , Patient-Centered Care
13.
Curr Opin Psychiatry ; 37(2): 57-64, 2024 03 01.
Article in English | MEDLINE | ID: mdl-38226540

ABSTRACT

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.


Subject(s)
Tic Disorders , Tourette Syndrome , Humans , Child , Tourette Syndrome/diagnosis , Tourette Syndrome/therapy , Tourette Syndrome/complications , Tic Disorders/complications , Tic Disorders/therapy , Behavior Therapy
14.
Article in English | MEDLINE | ID: mdl-38249547

ABSTRACT

Background: Tourette syndrome (TS) is a neurologic condition characterized by motor and phonic tics. Dystonic tics, including blepharospasm, are considered atypical or unusual in severe TS. Case Report: We report a severe case of TS with facial dystonic tics resembling blepharospasm in which the microlesion effect and a sustained therapeutic effect was observed with bilateral globus pallidus interna (GPi) deep brain stimulation (DBS). Discussion: Bilateral GPi DBS can be beneficial for blepharospasm-like tics and severe symptoms of TS. The improvements seen can be explained by the microlesion effect induced by DBS lead placement in the GPi.


Subject(s)
Blepharospasm , Tics , Tourette Syndrome , Humans , Tics/therapy , Blepharospasm/therapy , Globus Pallidus , Tourette Syndrome/therapy , Electrodes
15.
Clin Child Psychol Psychiatry ; 29(1): 301-311, 2024 Jan.
Article in English | MEDLINE | ID: mdl-37225162

ABSTRACT

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.


Subject(s)
Stuttering , Tics , Tourette Syndrome , Male , Child , Humans , Tourette Syndrome/complications , Tourette Syndrome/therapy , Tics/therapy , Tics/etiology , Stuttering/therapy , Stuttering/complications , Speech Therapy , Speech
16.
Clin Child Psychol Psychiatry ; 29(1): 287-300, 2024 Jan.
Article in English | MEDLINE | ID: mdl-37210660

ABSTRACT

BACKGROUND: The motor and vocal tics that characterise Tourette syndrome are stigmatizing and impact on quality of life. Behavioural interventions such as Exposure Response Prevention or Comprehensive Behavioural Interventions for Tics are first line treatment for Tourette syndrome, but availability is limited. This study is the first to explore the impact of an established manualised Exposure Response Prevention treatment protocol, developed for individual therapy, but here uniquely delivered intensively, to a group. METHODS: A naturalistic study comprised of a consecutive series of children (N = 20), aged 8-16 years (M = 12, SD = 2.17) were offered Exposure Response Prevention in one of two groups, delivered in series within a specialist clinic. Young people received the equivalent of 12 sessions (matching the manualised individual protocol). RESULTS: The YGTSS and Giles de la Tourette Syndrome Quality of Life Scale for Children and Adolescents (Satisfaction Scale) showed significant improvement following treatment with moderate to large effect sizes. Thirty-five percent of children demonstrated a reliable improvement on the YGTSS Global Tic Severity score. CONCLUSIONS: These data suggest an established Exposure Response Prevention protocol can be delivered in an intensive, group setting with a positive clinical outcome. Replication in a randomized controlled trial is an important next step.


Subject(s)
Tics , Tourette Syndrome , Adolescent , Child , Humans , Tourette Syndrome/therapy , Quality of Life , Feasibility Studies , Tics/therapy , Behavior Therapy/methods , Severity of Illness Index
17.
J Neurosurg ; 140(6): 1620-1629, 2024 Jun 01.
Article in English | MEDLINE | ID: mdl-38157543

ABSTRACT

OBJECTIVE: Deep brain stimulation (DBS) is an effective treatment for medically refractory Tourette syndrome (TS). Several effective targets have been reported, but there is still controversy about the networks involved in the efficacy of DBS for TS. Here, the authors aimed to identify the basal ganglia-thalamo-cortical networks associated with tic and obsessive-compulsive behavior (OCB) improvement and the network link between the two main targets for TS. METHODS: A retrospective analysis of 21 patients treated with pallidal and thalamic DBS was performed. Tics and OCB scores were recorded before and after DBS. The authors localized the electrodes in standard MNI (Montreal Neurological Institute) space and calculated the volume of tissue activated with the settings at the last follow-up to obtain areas of maximal improvement ("sweet spots") among all patients for the pallidal and thalamic targets. Tractography was used to show the white matter pathways associated with maximal tic and OCB improvement. RESULTS: Ten patients treated with pallidal DBS and 11 patients treated with thalamic DBS were included. Responder rates were 80% in the pallidal and 64% in the thalamic target groups. Sweet spots for tics and OCB clustered in several areas across the basal ganglia and thalamus delineated two main networks. Tic reduction in the pallidal target mapped to a limbic pallidothalamic network and in the thalamic target to the premotor thalamocortical network. Putting these two networks together will form the main output of the so-called limbic-motor interface network. However, OCB reduction mapped a dorsomedial prefrontal cortex/dorsal anterior cingulate (dmPFC/dACC) network. CONCLUSIONS: The authors demonstrated the involvement of the limbic-motor interface network during effective DBS for tics in patients with TS. OCB redution was associated with the additional involvement of dmPFC/dACC connections passing dorsal to the head of the globus pallidus pars externa on its way to the thalamus and midbrain.


Subject(s)
Deep Brain Stimulation , Tourette Syndrome , Humans , Tourette Syndrome/therapy , Tourette Syndrome/physiopathology , Deep Brain Stimulation/methods , Male , Female , Adult , Retrospective Studies , Young Adult , Adolescent , Treatment Outcome , Thalamus/diagnostic imaging , Globus Pallidus , Middle Aged , Limbic System/physiopathology , Limbic System/diagnostic imaging , Child , Nerve Net/diagnostic imaging , Nerve Net/physiopathology , Basal Ganglia/physiopathology , Neural Pathways/physiopathology , Obsessive-Compulsive Disorder/therapy , Obsessive-Compulsive Disorder/physiopathology
18.
Eur J Neurol ; 31(1): e16051, 2024 01.
Article in English | MEDLINE | ID: mdl-37644767

ABSTRACT

BACKGROUND AND PURPOSE: Very little is known about the long-term prognosis of patients with functional tic-like behaviours (FTLBs). We sought to characterize the trajectory of symptom severity over a 12-month period. METHODS: Patients with FTLBs were included in our prospective longitudinal child and adult clinical tic disorder registries at the University of Calgary. Patients were prospectively evaluated 6 and 12 months after their first clinical visit. Tic inventories and severity were measured with the Yale Global Tic Severity Scale (YGTSS). RESULTS: Eighty-three youths and adults with FTLBs were evaluated prospectively until April 2023. Mean YGTSS total tic severity scores were high at baseline, with a mean score of 29.8 points (95% confidence interval [CI] = 27.6-32.1). Fifty-eight participants were reevaluated at 6 months, and 32 participants were reevaluated at 12 months. The YGTSS total tic severity score decreased significantly from the first clinical visit to 6 months (raw mean difference = 8.9 points, 95% CI = 5.1-12.7, p < 0.0001), and from 6 to 12 months (raw mean difference = 6.4 points, 95% CI = 0.8-12.0, p = 0.01). Multivariable linear regression demonstrated that tic severity at initial presentation and the presence of other functional neurological symptoms were associated with higher YGTSS total tic scores at 6 months, whereas younger age at baseline, receiving cognitive behavioural therapy for anxiety and/or depression, and prescription of selective serotonin reuptake inhibitors were associated with lower YGTSS total tic scores at 6 months. CONCLUSIONS: We observed a meaningful improvement in tic severity scores in youth and adults with FTLBs over a period of 6-12 months.


Subject(s)
COVID-19 , Tic Disorders , Tics , Tourette Syndrome , Child , Adult , Humans , Adolescent , Follow-Up Studies , Pandemics , Prospective Studies , Severity of Illness Index , COVID-19/complications , Tic Disorders/epidemiology , Tic Disorders/therapy , Tic Disorders/complications , Tourette Syndrome/diagnosis , Tourette Syndrome/psychology , Tourette Syndrome/therapy
19.
Mov Disord ; 39(1): 183-191, 2024 Jan.
Article in English | MEDLINE | ID: mdl-38146055

ABSTRACT

BACKGROUND: Tourette syndrome (TS) tics are typically quantified using "paper and pencil" rating scales that are susceptible to factors that adversely impact validity. Video-based methods to more objectively quantify tics have been developed but are challenged by reliance on human raters and procedures that are resource intensive. Computer vision approaches that automate detection of atypical movements may be useful to apply to tic quantification. OBJECTIVE: The current proof-of-concept study applied a computer vision approach to train a supervised deep learning algorithm to detect eye tics in video, the most common tic type in patients with TS. METHODS: Videos (N = 54) of 11 adolescent patients with TS were rigorously coded by trained human raters to identify 1.5-second clips depicting "eye tic events" (N = 1775) and "non-tic events" (N = 3680). Clips were encoded into three-dimensional facial landmarks. Supervised deep learning was applied to processed data using random split and disjoint split regimens to simulate model validity under different conditions. RESULTS: Area under receiver operating characteristic curve was 0.89 for the random split regimen, indicating high accuracy in the algorithm's ability to properly classify eye tic vs. non-eye tic movements. Area under receiver operating characteristic curve was 0.74 for the disjoint split regimen, suggesting that algorithm generalizability is more limited when trained on a small patient sample. CONCLUSIONS: The algorithm was successful in detecting eye tics in unseen validation sets. Automated tic detection from video is a promising approach for tic quantification that may have future utility in TS screening, diagnostics, and treatment outcome measurement. © 2023 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.


Subject(s)
Deep Learning , Movement Disorders , Tic Disorders , Tics , Tourette Syndrome , Adolescent , Humans , Tics/diagnosis , Tic Disorders/diagnosis , Tourette Syndrome/diagnosis , Tourette Syndrome/therapy , Treatment Outcome
20.
J Clin Sleep Med ; 19(12): 2149-2151, 2023 12 01.
Article in English | MEDLINE | ID: mdl-38041536

ABSTRACT

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.


Subject(s)
Deep Brain Stimulation , Sleep Wake Disorders , Tourette Syndrome , Adolescent , Child , Humans , Artifacts , Tourette Syndrome/complications , Tourette Syndrome/therapy
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