Development and Open Trial of a Psychosocial Intervention for Young Children With Chronic Tics: The CBIT-JR Study

The Comprehensive Behavioral Intervention for Tic Disorders (CBIT) has demonstrated efficacy in large randomized controlled trials for children (≥9 yrs), adolescents and adults with Tourette Syndrome and Chronic Tic Disorders. Given the early age of onset for tic disorders, a large portion of affected individuals with chronic tic disorders are less than 9 years of age and appropriate developmental adaptations of behavioral treatment have not yet been tested. The goal of this study was to adapt and evaluate the acceptability and utility of a family-based adaptation of CBIT for children under 9 years of age. Children 5–8 years of age (N = 15) with chronic tics were recruited from three study sites. CBIT was adapted for use with young children and included habit reversal strategies introduced in a developmentally appropriate game format and function-based interventions to reduce family accommodation of and attention to tic symptoms.Children and parents described high level of treatment satisfaction and study retention rate was 100%. Treatment response rate was 54% (CGI-I = 1 or 2) with a significant decrease in the YGTSS total score (Cohen’s d = 0.73) that was largely maintained at 3-month and 1-year follow-up assessments. Treatment was associated with reduction of some symptoms of tic-related comorbid syndromes and with changes in parental accommodation and attention to tics.Future research should determine if parental attention to tics and symptom accommodation are important mediators of treatment outcome, or if participating in this intervention at a younger age may prevent the chronic course of tic symptoms.     

An Empirical Examination of Symptom Substitution Associated With Behavior Therapy for Tourette's Disorder

Over the past six decades, behavior therapy has been a major contributor to the development of evidence-based psychotherapy treatments. However, a long-standing concern with behavior therapy among many nonbehavioral clinicians has been the potential risk for symptom substitution. Few studies have been conducted to evaluate symptom substitution in response to behavioral treatments, largely due to measurement and definitional challenges associated with treated psychiatric symptoms. Given the overt motor and vocal tics associated with Tourette’s disorder, it presents an excellent opportunity to empirically evaluate the potential risk for symptom substitution associated with behavior therapy. The present study examined the possible presence of symptom substitution using four methods: (a) the onset of new tic symptoms, (b) the occurrence of adverse events, (c) change in tic medications, and (d) worsening of co-occurring psychiatric symptoms. Two hundred twenty-eight participants with Tourette’s disorder or persistent motor or vocal tic disorders were randomly assigned to receive behavioral therapy or supportive therapy for tics. Both therapies consisted of eight sessions over 10 weeks. Results indicated that participants treated with behavior therapy were not more likely to have an onset of new tic symptoms, experience adverse events, increase tic medications, or have an exacerbation in co-occurring psychiatric symptoms relative to participants treated with supportive therapy. Further analysis suggested that the emergence of new tics was attributed with the normal waxing and waning nature of Tourette’s disorder. Findings provide empirical support to counter the long-standing concern of symptom substitution in response to behavior therapy for individuals with Tourette's disorder.     

Executive and Attention Functioning Among Children in the PANDAS Subgroup

Evidence from past studies indicates that adults and children with Obsessive-Compulsive Disorder (OCD) and Tourette syndrome (TS) experience subtle neuropsychological deficits. Less is known about neuropsychological functioning of children and adolescents with a symptom course consistent with the PANDAS (Pediatric Autoimmune Neuropsychiatric Disorders Associated with Streptococcal infection) subgroup of OCD and tics. To provide such information, we administered three tests of attention control and two of executive function to 67 children and adolescents (ages 5–16) diagnosed with OCD and/or tics and a symptom course consistent with the PANDAS subgroup and 98 healthy volunteers (HV) matched by age, sex, and IQ. In a paired comparison of the two groups, the PANDAS subjects were less accurate than HV in a test of response suppression. Further, in a two-step linear regression analysis of the PANDAS group in which clinical variables were added stepwise into the model and in the second step matching variables (age, sex, and IQ) were added, IQ emerged as a predictor of performance on this task. In the same analysis, ADHD diagnosis and age emerged as predictors of response time in a continuous performance task. Subdividing the PANDAS group by primary psychiatric diagnosis revealed that subjects with TS or OCD with tics exhibited a longer response time compared to controls than subjects with OCD only, replicating previous findings within TS and OCD. This study demonstrates that children with PANDAS exhibit neuropsychological profiles similar to those of their primary psychiatric diagnosis.     

Obsessive-compulsive disorder with and without tic disorder: a comparative study from India

Introduction: Evidence from phenomenological, family, genetic, and treatment studies from Western centers have suggested that tic-related obsessive-compulsive disorder (OCD) could be different from non-tic-related OCD. This study from India investigated the differences in OCD with and without tics, with respect to sociodemographics, symptom profile, and comorbidity, including obsessive-compulsive spectrum disorders, to examine whether the clinical profile of tic-related OCD is similar to that reported previously. Methods: Fifty subjects with OCD and tics (chronic motor tics and Tourette syndrome) were compared with 141 OCD subjects without tics. Results: Subjects having OCD with tics tended to be males, and had an earlier onset of illness. They had more of symmetry/aggressive and religious obsessions, and cleaning, ordering/arranging, hoarding, and repeating compulsions and were associated with trichotillomania and hypochondraisis. Stepwise backward (Wald) regression analysis showed that an early age of onset, male gender, aggressive obsessions, cleaning compulsions, and trichotillomania were significantly associated with tic-related OCD. Conclusion: The findings of this study from India are broadly similar to those reported previously from the West indicating the universality of differences in tic- and non-tic-related OCD. Our findings also support the existing evidence that tics contribute to the heterogeneity of OCD.     

Ambulatory accelerometry to quantify involuntary movements and tics in the syndrome of Gilles de la Tourette

Gilles de la Tourette syndrome is a chronic neuropsychiatric disorder characterizedby recurrent and involuntary tics, in addition to complex behavioral symptoms. Objective quantification of the nonspecific movements in Tourette patients can contribute much to understanding the pathophysiology of this disease. We used three accelerometersto characterizehead movement patterns and to objectively quantify head motility in the lateral, sagittal, and transversal planes in 9 Tourette patients and 14 controls during periods of rest, conversation, and watching a videotape with an entertaining program. Characteristic head movement patterns can be documented by means of accelerometry. Head motility levels in the lateral, sagittal, and transversal planes were significantly higher in the patients than in the controls during all the procedures. The patients and the controls showed a similar significant increase in head motility during conversation, but not during video watching. This first study shows that for both standardized and ambulatory research, accelerometry may provide an objective tool by which to quantify the severity and temporal dynamics of tics or nonspecific movements.     

Treatment of vocal tics in children with Tourette syndrome: investigating the efficacy of habit reversal

Habit reversal was used to treat vocal tics in 5 children with Tourette syndrome. Vocal tics were reduced in 4 of the 5 children, the untreated motor tics did not increase, and treatment was acceptable to the children's parents.     

Moral Responsibility and Tourette Syndrome1

Philosophers generally assume that individuals with Tourette syndrome are not responsible for their Tourettic tics, and so not blameworthy for any harm their tics might cause. Yet this assumption is based largely on ignorance of the lived experience of Tourette syndrome. Individuals with Tourette syndrome often experience their tics as freely chosen and reason‐responsive. Yet it still seems wrong to treat a Tourettic individual's tic as on a moral par with others’ actions. In this paper, I examine the options and argue that, if this is correct, then a surprising consequence follows: the standard, motivation‐based theory of desire must be false. I go on to argue that, given what is known about the neurological basis of Tourette syndrome, this is a reasonable conclusion to draw.     

Habit reversal versus supportive psychotherapy in Tourette's disorder: a randomized controlled trial and predictors of treatment response

Tourette syndrome (TS) is characterized by chronic motor and vocal tics. Habit reversal therapy (HR) is a behavioral treatment for tics which has received recent empirical support. The present study compared the efficacy of HRT in reducing tics, improving life-satisfaction and psychosocial functioning in comparison with supportive psychotherapy (SP) in outpatients with TS. In addition, we investigated whether impairments in response inhibition in patients with TS predict response to HR treatment which specifically aims to inhibit tics. Thirty adult outpatients with DSM-IV TS were randomized to 14 individual sessions of HR (n = 15); or SP (n = 15). HR but not SP reduced tic severity over the course of the treatment. Both groups improved in life-satisfaction and psychosocial functioning during active treatment. Reductions in tic severity (HR) and improvements in life-satisfaction and psychosocial functioning (HR and SP) remained stable at the 6-month follow-up. The extent of pre-treatment response inhibition impairment in the HR group predicted reductions in tic-severity from pre- to post-treatment. Our results suggest that HR has specific tic-reducing effects although SP is effective in improving life-satisfaction and psychosocial functioning. Assessments of response inhibition may be of value for predicting treatment response to HR.     

A double-blind,sham-controlled,trial of home-administered rhythmic 10-Hz median nerve stimulation for the reduction of tics,and suppression of the urge-to-tic,in individuals with Tourette syndrome and chronic tic disorder

Tourette syndrome (TS) and chronic tic disorder (CTD) are neurological disorders of childhood onset characterized by the occurrence of tics; repetitive, purposeless, movements or vocalizations of short duration which can occur many times throughout a day. Currently, effective treatment for tic disorders is an area of considerable unmet clinical need. We aimed to evaluate the efficacy of a home-administered neuromodulation treatment for tics involving the delivery of rhythmic pulse trains of median nerve stimulation (MNS) delivered via a wearable ‘watch-like’ device worn at the wrist. We conducted a UK-wide parallel double-blind sham-controlled trial for the reduction of tics in individuals with tic disorder. The device was programmed to deliver rhythmic (10 Hz) trains of low-intensity (1–19 mA) electrical stimulation to the median nerve for a pre-determined duration each day, and was intended to be used by each participant in their home once each day, 5 days each week, for a period of 4 weeks. Between 18th March 2022 and 26th September 2022, 135 participants (45 per group) were initially allocated, using stratified randomization, to one of the following groups; active stimulation; sham stimulation or to a waitlist (i.e. treatment as usual) control group. Recruited participants were individuals with confirmed or suspected TS/CTD aged 12 years of age or upward with moderate to severe tics. Researchers involved in the collection or processing of measurement outcomes and assessing the outcomes, as well as participants in the active and sham groups and their legal guardians were all blind to the group allocation. The primary outcome measure used to assess the ‘offline’ or treatment effect of stimulation was the Yale Global Tic Severity Scale–Total Tic Severity Score (YGTSS–TTSS) assessed at the conclusion of 4 weeks of stimulation. The primary outcome measure used to assess the ‘online’ effects of stimulation was tic frequency, measured as the number of tics per minute (TPM) observed, based upon blind analysis of daily video recordings obtained while stimulation was delivered. The results demonstrated that after 4-week stimulation, tic severity (YGTSS-TTSS) had reduced by 7.1 points (35 percentile reduction) for the active stimulation group compared to 2.13/2.11 points for the sham stimulation and waitlist control groups. The reduction in YGTSS–TTSS for the active stimulation group was substantially larger, clinically meaningful (effect size = .5) and statistically significant (p = .02) compared to both the sham stimulation and waitlist control groups, which did not differ from one another (effect size = −.03). Furthermore, blind analyses of video recordings demonstrated that tic frequency (tics per minute) reduced substantially (−15.6 TPM) during active stimulation compared to sham stimulation (−7.7 TPM). This difference represents a statistically significant (p < .03) and clinically meaningful reduction in tic frequency (>25 percentile reduction: effect size = .3). These findings indicate that home-administered rhythmic MNS delivered through a wearable wrist-worn device has the potential to be an effective community-based treatment for tic disorders.     

The Premonitory Urge to Tic: Measurement,Characteristics, and Correlates in Older Adolescents and Adults

In addition to motor and/or vocal tics, many individuals with Tourette syndrome (TS) or chronic tic disorder (CTD) report frequent, uncomfortable sensory phenomena that immediately precede the tics. To date, examination of these premonitory sensations or urges has been limited by inconsistent assessment tools. In this paper, we examine the psychometric properties of a nine-item self-report measure, the Premonitory Urge to Tic Scale (PUTS) and examine the characteristics and correlates of the premonitory urge to tic in a clinical sample of 122 older adolescents and adults with TS or CTD. The PUTS demonstrated adequate internal consistency, temporal stability, and concurrent validity. Premonitory urges were endorsed by the majority of individuals. Most individuals reported some relief from the urges after completing a tic and being able to stop their tics even if only temporarily. Degree of premonitory urges was not significantly correlated with age, and we did not observe any gender differences. Degree of premonitory urges was significantly correlated with estimated IQ and tic severity, but not severity of comorbid obsessive–compulsive disorder or attention-deficit hyperactivity disorder. Also, it was not related to concomitant medication status. These findings represent another step forward in our understanding of the premonitory sensations associated with TS and CTD.     

Évaluation de l’effet d’un programme d’entraînement à l’autogestion des épisodes explosifs chez des enfants atteints du syndrome de Gilles de la Tourette

For children with Tourette syndrome (TS), explosive outbursts (EO) can be more disruptive than tics to the child's functioning. This study evaluated the effectiveness of an innovative cognitive-behavioral intervention for decreasing frequency and intensity of EO. Five boys and one girl ranging in age from 9 to 12 years took part in the study. EO frequency and intensity were measured by systematic observation of events and by questionnaire, while tics and child's psychosocial functioning were measured by questionnaires. The children showed trends towards decreased EO frequency post treatment but no changes in EO intensity. Statistical analysis and questionnaires results revealed no significant change. Qualitative information indicated a benefit to the families following therapy.     

The role of the cingulate cortex in the generation of motor tics and the experience of the premonitory urge-to-tic in Tourette syndrome

Tourette syndrome (TS) is a neurological disorder of childhood onset that is characterized by the occurrence of motor and vocal tics. TS is associated with cortical-striatal-thalamic-cortical circuit [CSTC] dysfunction and hyper-excitability of cortical limbic and motor regions that are thought to lead to the occurrence of tics. Individuals with TS often report that their tics are preceded by ‘premonitory sensory/urge phenomena’ (PU) that are described as uncomfortable bodily sensations that precede the execution of a tic and are experienced as a strong urge for motor discharge. While the precise role played by PU in the occurrence of tics is largely unknown, they are nonetheless of considerable theoretical and clinical importance as they form a core component of many behavioural therapies used in the treatment of tic disorders. Recent evidence indicates that the cingulate cortex may play an important role in the generation of PU in TS, and in ‘urges-for-action’ more generally. In the current study, we utilized voxel-based morphometry (VBM) techniques, together with ‘seed-to-voxel’ structural covariance network (SCN) mapping, to investigate the putative role played by the cingulate cortex in the generation of motor tics and the experience of PU in a relatively large group of young people with TS. Whole-brain VBM analysis revealed that TS was associated with clusters of significantly reduced grey matter volumes bilaterally within: the orbito-frontal cortex; the cerebellum; and the anterior and mid-cingulate cortex. Similarly, analysis of SCNs associated with bilateral mid- and anterior cingulate ‘seed’ regions demonstrated that TS is associated with increased structural covariance primarily with the bilateral motor cerebellum; the inferior frontal cortex; and the posterior cingulate cortex.     

Motor excitability is reduced prior to voluntary movements in children and adolescents with Tourette syndrome

Tourette syndrome (TS) is a neuro‐developmental disorder characterized by the occurrence of motor and vocal tics: involuntary, repetitive, stereotyped behaviours that occur with a limited duration, often typically many times in a single day. Previous studies suggest that children and adolescents with TS may undergo compensatory, neuroplastic changes in brain structure and function that help them gain control over their tics. In the current study we used single‐pulse and dual‐site paired‐pulse transcranial magnetic stimulation (TMS), in conjunction with a manual choice reaction time task that induces high levels of inter‐manual conflict, to investigate this conjecture in a group of children and adolescents with TS, but without co‐morbid Attention Deficit Hyperactivity Disorder (ADHD). We found that performance on the behavioural response‐conflict task did not differ between the adolescents with TS and a group of age‐matched typically developing individuals. By contrast, our study demonstrated that cortical excitability, as measured by TMS‐induced motor‐evoked potentials (MEPs), was significantly reduced in the TS group in the period immediately preceding a finger movement. This effect is interpreted as consistent with previous suggestions that the cortical hyper‐excitability that may give rise to tics in TS is actively suppressed by cognitive control mechanisms. Finally, we found no reliable evidence for altered patterns of functional inter‐hemispheric connectivity in TS. These results provide evidence for compensatory brain reorganization that may underlie the increased self‐regulation mechanisms that have been hypothesized to bring about the control of tics during adolescence.     

Hoarding in obsessive compulsive disorder: results from a case-control study

Hoarding occurs relatively frequently in obsessive-compulsive disorder (OCD), and there is evidence that patients with hoarding symptoms have more severe OCD and are less responsive to treatment. In the present study, we investigated hoarding symptoms in 126 subjects with OCD. Nearly 30% of the subjects had hoarding symptoms; hoarding was twice as prevalent in males than females. Compared to the 90 non-hoarding subjects, the 36 hoarding individuals had an earlier age at onset of, and more severe, obsessive-compulsive symptoms. Hoarders had greater prevalences of symmetry obsessions, counting compulsions, and ordering compulsions. Hoarders also had greater prevalences of social phobia, personality disorders, and pathological grooming behaviors (skin picking, nail biting, and trichotillomania). Hoarding and tics were more frequent in first-degree relatives of hoarding than non-hoarding probands. The findings suggest that the treatment of OCD patients with hoarding symptoms may be complicated by more severe OCD and the presence of co-occurring disorders. Hoarding appears to be transmitted in some OCD families and may differentiate a clinical subgroup of OCD.     

Functional analysis and habit‐reversal treatment of tics

Although there is empirical support for a biological etiology of Tourette syndrome and other tic disorders, researchers have recently begun examining the role of reinforcement in tic maintenance. Using experimental functional analyses, researchers have identified both social and nonsocial functions for the tics of a variety of individuals. However, there are at least two problems with this developing literature. First, all but one of the studies employed a single participant and many of them were referred for clinical treatment. These factors leave open the possibility that the selection or referral process may have contributed to the reported outcomes. Second, five of the seven participants had a developmental disability or delay. Thus, the majority of participants in this literature are unrepresentative of most individuals with tic disorders. The purpose of the present study was to investigate the operant functions of the tics of multiple children who were recruited for the study. We evaluated the tics of three typically developing children using functional analysis. Results indicated nonsocial functions for all tics. Two of the participants were then treated using habit reversal; both of them experienced tic reductions. Copyright © 2005 John Wiley & Sons, Ltd.     

THE ROLE OF INDEPENDENT VARIABLE INTEGRITY IN THE BEHAVIORAL MANAGEMENT OF TOURETTE SYNDROME

We examined the effects of self-monitoring, dissimilar response practice, and competing response practice on the tics of two preadolescent males with Tourette Syndrome. Tic frequencies were assessed using an alternating treatments design over the course of approximately 1 month. The data indicate that neither of the participants’ tics changed as a result of the interventions. Upon examination of independent variable integrity data, we noticed that the participants were not implementing the treatments as instructed. The results are discussed in the context of improving the research methodology in this area, particularly with respect to independent variable integrity measures.     

Durability,Negative Impact,and Neuropsychological Predictors of Tic Suppression in Children with Chronic Tic Disorder

Chronic tic disorders are characterized by involuntary motor and vocal tics, which are influenced by contextual factors. Recent research has shown that (a) children can suppress tics for brief periods of time, (b) suppression is enhanced when programmed reinforcement is provided for tic-free intervals, and (c) short periods of suppression do not result in a paradoxical "rebound" in tic frequency when active suppression has ceased. The current study extended existing research in three important ways. First, we examined whether tic suppression ability decreased as suppression duration increased from 5 to 25 to 40 min. Second, we examined post-suppression tic frequency to test whether longer periods of suppression were more likely to be associated with a rebound effect. Finally, we explored neuropsychological predictors of tic suppression. Thirteen children with Tourette syndrome or a chronic tic disorder completed the study. Results showed that (a) tic suppression was sustained for all of the suppression durations, (b) rebound effects were not observed following any of the suppression durations, and (c) ability to suppress was correlated with omission, but not commission errors on a continuous performance task. Implications of these findings are discussed.     

Motor excitability during movement preparation in Tourette syndrome

Tourette syndrome (TS) is a neurodevelopmental disorder characterized by the occurrence of motor and vocal tics. TS has been linked to the impaired operation of cortical‐striatal‐thalamic‐cortical circuits that give rise to hyper‐excitability of cortical motor areas, which may be exacerbated by dysfunctional intra‐cortical inhibitory mechanisms. That said, many individuals gain control over their tics during adolescence and it has been suggested that this increased control arises as a result of the development of mechanisms that operate to suppress corticospinal excitability (CSE) ahead of volitional movements. Here we used single‐pulse transcranial magnetic stimulation (TMS) in conjunction with a manual Go/NoGo task to investigate alterations in CSE ahead of volitional movements in a group of adolescents with TS (N = 10). Our study demonstrated that CSE, as measured by TMS‐induced motor‐evoked potentials (MEPs), was significantly reduced in the TS group in the period immediately preceding a finger movement. More specifically, we show that individuals with TS, unlike their age‐matched controls, do not exhibit the predicted increase in mean MEP amplitude and decrease in MEP variability that immediately precede the execution of volitional movements in typically developing young adults. Finally, we report that the magnitude of the rise in MEP amplitude across the movement preparation period in TS is significantly negatively correlated with clinical measures of motor tic severity, suggesting that individuals with severe motor tics are least able to modulate motor cortical excitability.     

Premonitory urges are associated with decreased grey matter thickness within the insula and sensorimotor cortex in young people with Tourette syndrome

Tourette syndrome (TS) is a neurological disorder characterized by vocal and motor tics and is associated with cortical–striatal–thalamic–cortical circuit (CSTC) dysfunction and hyperexcitability of cortical limbic and motor regions, which are thought to lead to the occurrence of tics. Importantly, individuals with TS often report that their tics are preceded by ‘premonitory sensory phenomena’ (PSP) that are described as uncomfortable cognitive or bodily sensations that precede the execution of a tic, and are experienced as a strong urge for motor discharge. While the precise role played by PSP in the occurrence of tics is controversial, PSP are nonetheless of considerable theoretical and clinical importance in TS, not least because they form the core component in many of the behavioural therapies that are currently used in the treatment of tic disorders. In this study, we investigated the brain structure correlates of PSP. Specifically, we conducted a whole‐brain analysis of cortical (grey matter) thickness in 29 children and young adults with TS and investigated the association between grey matter thickness and PSP. We demonstrate for the first time that PSP are inversely associated with grey matter thickness measurements within the insula and sensorimotor cortex. We also demonstrate that grey matter thickness is significantly reduced in these areas in individuals with TS relative to a closely age‐ and gender‐matched group of typically developing individuals and that PSP ratings are significantly correlated with tic severity.     

The development of stimulus control over tics: A potential explanation for contextually-based variability in the symptoms of Tourette syndrome

Research has demonstrated that providing reinforcement for tic-free intervals can decrease tic frequency in controlled analogue settings. The aim of the current study was to determine whether reinforcement could be used to create stimulus control over tic expression. Ten children with chronic tic disorders (including Tourette syndrome) completed four discrimination training sessions. Each session consisted of three exposures to each of three, 5 min conditions presented in a random order. In one condition, participants were reinforced for tic absence on a 10-s fixed interval schedule in the presence of a purple light. In a second condition, participants were instructed to suppress their tics, but were not reinforced for doing so in the presence of an orange light. In a third condition, participants were instructed not to suppress their tics in the presence of two non-illuminated lights. Confirming findings from other studies, results showed that reinforcing tic suppression reduced tic frequency to a greater extent than only providing instructions to suppress. To test for stimulus control, a fifth session was conducted following the aforementioned discrimination training sessions. The fifth session consisted of three exposures to each of three 5 min conditions presented in a random order. In one condition, a purple light was illuminated. In a second condition, an orange light was illuminated. In a third condition, neither light was illuminated. Across all three conditions, instructions to suppress (or not suppress) tics were not provided, and reinforcers for successful suppression were not delivered. Results indicated that in the presence of the purple light, tics were significantly lower than when neither light was illuminated. These findings provide preliminary support for the idea that a history of differential reinforcement in various contexts may play a role explaining variability in tic symptom expression.