Description,Implementation,and Efficacy of the Comprehensive Behavioral Intervention for Tics as First-Line Treatment for Tourette and Other Tic Disorders

Abstract

Objective:

To provide an evidence-based review of the Comprehensive Behavioral Intervention for Tic (CBIT) disorders.

Results:

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

Conclusions:

CBIT has demonstrated acute and durable efficacy when delivered alone or in combination with medication, in person, or via telehealth, and in the presence or absence of common comorbid conditions. Additional research is needed to develop and test treatment guidelines for the use of CBIT in combination with pharmacologic, neuromodulatory, and other intervention modalities.

Introduction

As our understanding of Tourette and other tic disorders (TDs) has evolved, so too have the recommendations for treating these conditions. While medication remains an important part of TD treatment for many individuals, there has been a notable shift in the recommendations for first-line treatment of TD from pharmacological to behavioral interventions (Capriotti et al., 2014; Pringsheim et al., 2019a; Steeves et al., 2012). This shift is based, at least in part, on the understanding that tics can be suppressed for different lengths of time (Himle and Woods, 2005; Woods and Himle, 2004) and can be impacted by contextual factors, including internal (e.g., premonitory urge, emotional states) and external (e.g., specific situations/activities, social responses, task avoidance) triggers (Capriotti et al., 2014; Himle et al., 2006; Woods et al., 2008). While tics are neurological in nature, they are sensitive to the environments, both internal and external, in which they occur (Piacentini et al., 2007). Recognizing that individuals with TD can exert some control over these environmental triggers, behavioral interventions represent an important avenue through which individuals can receive targeted symptom relief without the potential burden of medication side effects or as an adjunct to pharmacotherapy. The aim of this article is to describe the Comprehensive Behavioral Intervention for Tics (CBIT), an individualized, multicomponent treatment approach to treating TD (Woods et al., 2008). CBIT has demonstrated efficacy for reducing tic severity in youth and adults (Piacentini et al., 2010; Wilhelm et al., 2012), with treatment gains in youth maintained for up to 11 years posttreatment (Espil et al., 2022).

Habit Reversal Training as a Precursor to CBIT

The primary component of and precursor to CBIT is habit reversal training (HRT). HRT was the first multimodule behavioral intervention package that sought to treat tics and other habit-based disorders (e.g., thumb sucking, trichotillomania). As originally described by Azrin and Nunn (1973), HRT consists of psychosocial and behavioral skills that work to increase awareness of tics, provide competing responses to the behaviors, and increase social support to encourage ongoing practice of these skills. HRT was predicated on the conceptualization of “nervous habits” (including tics) as behaviors maintained by response chains that needed to be interrupted and further perpetuated by a lack of awareness, excessive repetition, social reinforcement (e.g., sympathy), and tolerance for their occurrence (Azrin and Nunn, 1973). More specifically, by repeatedly blocking tic suppression in the presence of the premonitory urge, both urge and tic would eventually disappear.

Although HRT originally involved 14 separate components, subsequent research has identified three specific skills as the primary active components for achieving tic management: awareness training, competing response training, and social support (Miltenberger et al., 1985; Woods et al., 1996). In addition, given that anxiety and stress have been shown to exacerbate tics, relaxation training has historically been included in HRT (Azrin and Nunn, 1973).

The first component of HRT, tic awareness training, is designed to increase early recognition of the targeted tic as well as the premonitory urge or other signals (e.g., muscle tightening) that tic expression is imminent (Piacentini and Chang, 2005). Patients are taught to identify each time the tic is expressed (e.g., bodily location, muscles involved, sound made), the contexts in which the tic tends to emerge, the frequency of the tic’s occurrence, and the premonitory urges and sensations that immediately precede the tic’s expression. Increased awareness facilitates implementation of a competing response, or a voluntary behavior that is physically incompatible with the targeted tic’s expression. The competing response should also be socially discrete, and able to be maintained for sustained periods of time (at least a minute or until tic expression is no longer imminent) and in various contexts (Azrin and Nunn, 1973; Piacentini et al., 2010). Sequentially, once an urge to tic arises, the competing response is used to inhibit expression of the associated tic thereby disrupting the potential reinforcing aspects of the tic as a means of resolving the discomfort associated with the premonitory urge.

Finally, implementation of competing response training is practiced in the patient’s natural environment, initially with an identified caregiver or support person who can encourage the patient to use these skills and provide praise and support for doing so. When implemented consistently and correctly, HRT can be an effective tool for disrupting the automatized “urge–tic–relief” cycle thought to maintain tics. As with any behavioral intervention, regular practice is important to generalize tic management across relevant settings and maximize the durability of treatment gains over time (McGuire et al., 2020a).

In the first controlled study of HRT for tics, active treatment led to a significant reduction in tic frequency in all 10 participants (ages 8–36), with 9 of 10 individuals showing at least 88% tic reduction in both the home and clinic settings (Azrin and Peterson, 1990). HRT also led to a significantly greater reduction in tic severity than supportive psychotherapy in a randomized trial of adults with TD, with HRT-related gains maintained at 6 months posttreatment (Deckersbach et al., 2006). HRT has been combined with a range of psychosocial interventions, including acceptance and commitment therapy (ACT), sleep hygiene, mindfulness, and physical exercise, with the goal of increasing treatment efficacy and durability, and enhancing quality of life (McGuire et al., 2020b).

Development of CBIT

Although HRT was regarded as a front-line behavioral treatment for tics from the initial publication of the Azrin and Nunn (1973) study through the early 2000s, the use of HRT and other behavioral techniques was greatly limited by the lack of trained providers, the absence of larger scale, well-controlled trials, and the widely accepted misconception that, given their neurobiological underpinnings, tics were fully involuntary and not amenable to behavioral intervention (Barnea et al., 2016; Himle and Woods, 2005; Peterson and Azrin, 1992; Piacentini et al., 2006; Piacentini and Chang, 2005).

The development of CBIT began in the early 2000s when the Tourette Association of America (TAA) convened a Behavioral Science Consortium (BSC) to develop and test a “promising behavioral treatment” that could serve as an alternative or supplement to pharmacological interventions for TD (Woods et al., 2008). The BSC included clinicians, scientists, and other experts in the field from various sites across the country, including the University of California-Los Angeles, University of Wisconsin-Milwaukee, Johns Hopkins University, Yale University of Texas-San Antonio, and Harvard/Massachusetts General Hospital. Following a comprehensive review of the extant literature, the BSC selected HRT and functional intervention as the most promising behavioral techniques for reducing tic severity and impairment, and combined these treatments into a single, manualized intervention, namely CBIT.

Once manualized, CBIT was tested in two large NIMH/NINDS-funded multisite randomized controlled trials comparing the treatment to psychoeducation with supportive therapy (PST) in youth aged 9–17 years (Piacentini et al., 2010) and adults aged 16–69 years (Wilhelm et al., 2012) with TD. In both studies, participants were randomly assigned to eight sessions of CBIT or PST over 10 weeks, with outcome data collected at posttreatment and follow-up visits 3 and 6 months posttreatment. Compared with PST, CBIT proved superior in tic reduction on the Yale Global Tic Severity Scale (YGTSS) severity score for both youth (d = 0.68) and adults (d = 0.57), although participants in the child/adolescent study showed a larger overall response rate (52.5%) on the Clinical Global Impressions-Improvement Scale (CGI-I) than adult participants (38.1%). The authors theorized that this developmental difference could be related to the significantly longer chronicity of TDs in the older group (Wilhelm et al., 2012). The effect size for the youth study was broadly comparable with that found for controlled pediatric trials of antipsychotic medications, especially when considering the active versus placebo comparison groups used in CBIT and the medication trials, respectively (Sallee et al., 2009; Scahill et al., 2003). Relatedly, for youth, but not adults, CBIT also outperformed PST in reducing tic-related impairment (Piacentini et al., 2010; Wilhelm et al., 2012). Both youth and adult trials demonstrated low attrition rates and did not identify any adverse events related to the intervention. Collectively, these trials established CBIT as a safe, well-tolerated, and efficacious treatment for reducing tic severity and, in some cases, improving global outcomes across a broad developmental spectrum.

The effects of CBIT have extended beyond tic reduction to other psychiatric and psychosocial outcomes. Using data from the youth CBIT trial, Woods and colleagues (2011) found that while immediate differences in co-occurring psychiatric symptoms were not noted at posttreatment, positive responders to CBIT at the 6-month follow-up demonstrated decreased anxiety, disruptive behavior, and family strain, as well as improved social functioning, compared with participants receiving PST. Findings suggest that CBIT may provide secondary gains in other non-TD psychiatric symptom domains once TD symptoms are addressed.

Long-Term Durability of CBIT

Although CBIT gains have been shown to persist 6 and 12 months posttreatment across numerous studies (Haas et al., 2022; Piacentini et al., 2010; Rachamim et al., 2022; Wilhelm et al., 2012), only one published study has assessed CBIT durabilty beyond this time frame (Espil et al., 2022). Using participants from the original youth CBIT randomized controlled trial (Piacentini et al., 2010), Espil and colleagues (2022) found that youth demonstrating an initial posttreatment positive response to CBIT were significantly more likely to be rated as responders (CGI-I = much or very much improved) compared with PST responders as well as CBIT and PST nonresponders 11 years after completing acute study treatment. In addition, initial CBIT responders had a significantly lower mean YGTSS total severity score than the initial PST responders at the 11-year follow-up (d = 1.47). No differences in YGTSS tic impairment scores between groups were noted, however, perhaps suggesting that individuals may become similarly acclimated to their tics over time despite experiencing different rates of improvement in tic severity (Espil et al., 2022; Groth et al., 2017). Notably, at the 11-year follow-up assessment, CBIT responders also had a higher occurrence of clinically meaningful improvement in tic severity (80.9%) compared with PST responders (50%), with 67% of CBIT responders meeting the threshold for at least partial remission (compared with 0% for PST responders). However, the small number of original PST responders in this sample somewhat limits interpretation.

Using the same follow-up sample, Ricketts and colleagues (2022) assessed the childhood predictors of adult tic severity and impairment, as well as changes in these outcomes over time. After controlling for prior tic treatment effects, these investigators also identified higher tic severity in childhood and female sex as predictors of higher tic severity and impairment in adulthood. In addition, poorer family functioning and the lack of stimulant medication use during childhood predicted higher tic impairment in adulthood (Ricketts et al., 2022). Regarding predictors of change over time, higher externalizing symptoms in childhood predicted greater reduction in both tic severity and impairment. Greater tic severity and premonitory urge severity in childhood predicted less improvement in tic severity over time, while female sex predicted smaller reduction in tic impairment (Ricketts et al., 2022).

Based on a systematic review of all available literature, the American Academy of Neurology (AAN) has designated CBIT as the first-line intervention for tics, when available, relative to other psychosocial/behavioral or psychopharmacological approaches (Pringsheim et al., 2019a). The European and Canadian guidelines also recommend CBIT or HRT as first-line treatments for Tourette and other TDs (Steeves et al., 2012; Verdellen et al., 2011).

Permutations of CBIT

Various adaptations to the original CBIT protocol have since been created to accommodate the need for flexibility in patient age (i.e., children vs. adults), delivery method (i.e., in-person vs. telehealth), therapist involvement (i.e., therapist delivered vs. caregiver guided), duration and intensity of treatment (i.e., an hour weekly for 2 months vs. 3–4 hours daily for 1 week), and practitioner expertise (i.e., psychologist vs. neurologist vs. occupational therapist).

Remote CBIT

Despite the robust support for the efficacy of CBIT in both youth (Piacentini et al., 2010) and adults (Wilhelm et al., 2012) with tics, many individuals seeking treatment do not have access to CBIT or other evidence-based interventions due to barriers, including the lack of trained providers and concerns about costs, distance, and time (Hollis et al., 2016; Woods et al., 2010). To address this gap, researchers have modified CBIT to allow for remote administration through platforms such as videoconferencing (Himle et al., 2010; Himle et al., 2012; Ricketts et al., 2016b) and internet-based programs (Haas et al., 2022; Rachamim et al., 2022). Further spurred by the COVID-19 lockdown, telehealth delivery of CBIT is now recognized as an effective modality for treating tics and comorbid disorders, with comparable benefits to face-to-face CBIT delivery (Andrén et al., 2019; Haas et al., 2022; Himle et al., 2010; Himle et al., 2012; Ricketts et al., 2016b).

Himle and colleagues’ initial feasibility study (2010) of remote CBIT through videoconferencing demonstrated significant tic reduction and high ratings of treatment acceptability and therapeutic relationship by both parents and children. In a larger study comparing remote with in-person CBIT, Himle and colleagues (2012) found that participants in both arms experienced significant tic reduction on the YGTSS, comparable in scope to the original controlled CBIT trial (Piacentini et al., 2010), and reported strong acceptability and therapist–client alliance, with no between-group differences on any outcome measures. Using a voice over internet protocol for CBIT, Ricketts and colleagues (2016b) found similar findings suggesting greater reductions in clinician-rated and parent-reported tics on the YGTSS and higher responder rates on the CGI-I for participants enrolled in CBIT compared with a waitlist control, thus generating further support for the efficacy of remote CBIT.

Internet-Delivered CBIT

To further address the accessibility treatment barriers, Rachamim and colleagues (2020; 2022) and Haas and colleagues (2022) evaluated the feasibility and effectiveness of an internet-delivered CBIT (ICBIT) adaptation for youth and adults, respectively, with chronic TDs. While ICBIT for youth used a caregiver-guided model, ICBIT for adults was entirely self-administered; importantly, neither ICBIT models required the supervision of a trained therapist. Results from the ICBIT for youth study (Rachamim et al., 2022) suggested strong acceptability and feasibility of the internet-delivered model, in addition to significant reductions in tic severity on the YGTSS from pre- to postintervention (d = 0.91), with an even larger effect 6 months posttreatment (d = 2.25). Effect sizes for the ICBIT youth adaptation were comparable with face-to-face delivery treatment trials (Piacentini et al., 2010). The adult ICBIT RCT (Haas et al., 2022) showed greater improvement in tic severity from pre- to posttreatment for participants in the ICBIT condition compared with the psychoeducation control group, with increased gains noted at the 3- and 6-month follow-ups. Participants in the ICBIT and face-to-face conditions showed similar gains, suggesting comparable benefits across delivery modalities.

Intensive outpatient CBIT

Blount and colleagues (Blount et al., 2014; Blount et al., 2018) compressed the weekly outpatient CBIT protocol into an intensive outpatient program (IOP) delivered 3–4 hours daily over the course of 1 week. Although there is a precedence for adapting multiweek/month treatment programs into the IOP structures in other disorders (e.g., Whiteside et al., 2008), research on the IOP CBIT adaptation is limited to two case series studies (Blount et al., 2014; Blount et al., 2018), involving a total of 7 participants with TD (four children and three young adults). While preliminary, findings from these two studies suggest a reduction in tic severity on the YGTSS and improvement on the CGI-I, with gains maintained at follow-up.

CBIT-JR

Given the early age of tic onset (typically around 5–7 years) (Gadow et al., 2002), Bennett and colleagues (Bennett et al., 2020) created a family-based adaptation of CBIT for children aged 5 to 8 years (CBIT-JR). CBIT-JR presents the manualized CBIT material in a developmentally appropriate game format with increased familial involvement. Preliminary findings from an open trial of 15 children suggest comparable reductions in tic severity to the original CBIT RCT (Piacentini et al., 2010), as well as decreases in family accommodations and improvements in non-tic psychiatric symptoms. Treatment gains were largely maintained at the 3-month and 1-year follow-up assessments.

CBIT-NP

CBIT has also been modified for use in child neurology and developmental pediatric clinics (CBIT-NP; Ricketts et al., 2016a), given these settings are often the first points of therapeutic contact for individuals with TD. In CBIT-NP, the frequency of treatment sessions was reduced from 8 sessions to 6, the duration of each session was reduced from 60–90 minutes to 20–25 minutes, and the content was delivered by a professional with expertise in neurology and/or developmental pediatrics. While preliminary, CBIT-NP showed significant reductions in clinician-rated tic severity and tic impairment on the YGTSS.

CBIT for occupational therapy

While effective in reducing the frequency and severity of tics, CBIT has also been found to improve self-rated occupational performance in children with TD (Rowe et al., 2013). Specifically, Rowe and colleagues found that once participants were better able to manage their tics through CBIT, they were able to focus more on valued activities (e.g., schoolwork, social activities, sleep) and to feel more confident in their ability to complete the necessary tasks; these gains, perhaps most importantly, resulted in participants ascribing greater importance to these activities in their everyday lives.

Group CBIT

Group-based CBIT represents a promising CBIT adaptation to combat the shortage of CBIT providers and to offer families a more cost-effective treatment option. In a randomized controlled trial comparing group-based CBIT (group-CBIT) to a group-based educational intervention for tics (group-EIT) (Zimmerman-Brenner et al., 2022), group-CBIT yielded stronger maintenance of observed reductions in motor, vocal, and total tic severity 3 months posttreatment. Importantly, these authors noted that the group-based interventions were more effective for children with higher intellectual ability and without significant behavioral challenges, highlighting the importance of understanding the child’s psychiatric and developmental profile in determining course of treatment. This study also identified additional benefits of group-CBIT on cognitive inhibition and cognitive reappraisal, although it is unclear if these improvements are unique to group-CBIT or if they apply to CBIT more generally (Gur et al., 2023). Kang et al. (2022) found group-CBIT to yield significantly greater improvement in tic-related interference, impairment, and global tic severity compared with a treatment as usual comparison condition.

Modified-CBIT

Recent efforts have been made to adapt CBIT to better address attention-deficit/hyperactivity disorder (ADHD) symptoms. In a 2023 pilot study, Greenberg and colleagues sought to evaluate the efficacy of a CBIT protocol that was modified to include ADHD-related treatment modules. Between-group comparisons found that CBIT and modified-CBIT (MCBIT) were equally as effective in reducing tic-related impairment and tic and ADHD symptoms severity overall. While group comparisons demonstrated the utility of an MCBIT protocol adapted for ADHD, the results of this study emphasize the utility of a flexibly implemented CBIT protocol, which may expand the clinical focus beyond addressing tics and still yield results.

CBIT and Concomitant Medication Use

As noted earlier, pharmacotherapy is recommended as a secondary treatment option to CBIT, when CBIT is available and not contraindicated by patient age, uncontrolled comorbidities, patient preference, or other potential complicating factors (Pringsheim et al., 2019a). However, combined treatment approaches (CBIT + medication) are common in many settings where both of these interventions are available. While analysis of the combined adult and child CBIT RCT samples identified concurrent tic suppressing medications as a significant moderator of response to CBIT, through which participants who were not on such medications demonstrated more robust treatment responses (Sukhodolsky et al., 2017), this effect appeared to be differentially carried by data from the adult treatment sample. In addition, the moderation effect may also have been driven by the type of tic-suppressing medication used or greater severity of the medication subgroup, although premedication baseline YGTSS scores for those who entered the trial on medication were unknown (Sukhodolsky et al., 2017). In our experience, combining CBIT with evidence-based pharmacological intervention (Pringsheim et al., 2019a) can be effective in many cases.

CBIT for Youth with TD and Comorbid Disorders

Approximately 85% of patients with Tourette syndrome (TS) experience other comorbid psychological, behavioral, or developmental disorders (Martino et al., 2017; Martino and Pringsheim, 2018). Among the most common are obsessive-compulsive disorder (OCD), ADHD, mood disorders, anxiety disorders, autism spectrum disorders (ASDs), and disruptive behavioral disorders (e.g., oppositional defiant and conduct disorders) (Martino et al., 2017; Pringsheim et al., 2019b). As many of the functional difficulties experienced by people with TS may be caused by co-occurring conditions, a thorough assessment of comorbidities should be conducted before any tic severity evaluations or intervention planning activities (Martino and Pringsheim, 2018).

Co-occurring OCD, ADHD, anxiety, mood, and/or disruptive disorders were present in the primary CBIT clinical trials study samples, although their inclusion was contingent upon the clinical stability of these symptoms in relation to TD (Piacentini et al., 2010; Wilhelm et al., 2012). Findings around the effects of ADHD on CBIT outcomes are equivocal and warrant further exploration (Andrén et al., 2022; Conelea and Wellen, 2017; Sukhodolsky et al., 2017). In their meta-analysis, McGuire and colleagues (2014) found that the CBIT response may be attenuated by symptoms of ADHD, although individuals with this comorbidity still exhibited comparable benefits to those without it. Conversely, Sukhodolsky and colleagues (2017) reported that ADHD did not moderate outcomes across the original CBIT combined child and adult samples, while Conelea and Wellen (2017) found youth (aged 5–17) with co-occurring ADHD to be as effective at suppressing tics as their non-ADHD counterparts. Collectively, these findings offer similar clinical conclusions that CBIT is not contraindicated in youth with relatively stable and managed ADHD, and that it is important that treatment of ADHD symptoms be initiated or optimized before the onset of CBIT (Greenberg et al., 2023).

It is estimated that a diagnosis of OCD may be present for 10–50% of youths with TD (Pringsheim et al., 2019b). Comorbid OCD appears to be associated with more severe tics and may put youths at greater risk of other psychopathology, such as depression and anxiety (Lebowitz et al., 2012).

Similar clinical profiles may initially make it difficult to differentiate between tics and OCD-related compulsions; thus, careful evaluation is an important aspect of assessment and treatment. Compulsions typically occur in response to a distressing thought or image, and often take much longer than tics to complete, whereas tics are associated with a premonitory urge and lack a cognitive “purpose” for their completion. Clinical experiences with this population indicate that with appropriate psychoeducation, many youths are able to readily distinguish between tics and compulsive urges, and the subsequent distress and impairment from them should be considered when deciding which symptoms to target first. Overall, CBIT appears to be as effective for youths with co-occurring OCD (McGuire et al., 2014; Sukhodolsky et al., 2017). Importantly, CBIT is recommended by AAN guidelines as the first-line treatment for tics that co-occur with OCD (Pringsheim et al., 2019b).

While less frequently co-occurring than ADHD, OCD, and other anxiety disorders (Scahill et al., 2014), youths with TD experience higher rates of mood disorders than those without (Pringsheim et al., 2019b). Notably, well-controlled mood disorder symptoms are represented in the CBIT literature and do not appear to moderate the effectiveness of the treatment protocol (McGuire et al., 2014; Sukhodolsky et al., 2017).

Experience with individuals with TD and mood disorders indicates that severely impairing tics may be a risk factor for the development and maintenance of depressive symptoms, self-injury, and suicide. Risk for suicide is reflected in the literature, with higher levels of risk noted among youths with more complex clinical presentations (e.g., severe tics, presence of comorbidities) (Storch et al., 2015). In addition, individuals with tics experience higher rates of bullying, lower quality of life, and worsened family functioning overall, which compounds the risk for the development of affective symptoms (Pringsheim et al., 2019b; Scahill et al., 2014; Vermilion et al., 2020).

TD is a common comorbidity in ASD, with prevalence rates ranging from 5% to 20% (Baron-Cohen et al., 1999; Canitano and Vivanti, 2007; Kadesjö and Gillberg, 2000; Kalyva et al., 2016; Simonoff et al., 2008). Although CBIT treatment studies for this population are yet to be published, clinical observations, including our own practices, suggest that CBIT may be a viable treatment option given the overlap in approaches seen across ASD and TD presentations. CBIT comprises various techniques that are commonly used in treatments for ASD-related symptoms, including a focus on external factors impacting behavior (Dargue et al., 2022), functional behavioral assessments (Vismara and Rogers, 2010), incorporating parent support and psychoeducation (Kasari and Lawton, 2010), and concrete strategies to use as a replacement for other behaviors, among others. Thus, clinically, CBIT may lend itself well to this population with some modifications to provide additional structure and support. Various potential treatment adaptations have been suggested for youth with TD and comorbidities generally (Bolen et al., 2022), although there is a dearth of literature on outcomes and adaptations when ASD is the comorbidity.

CBIT-Relevant Myths and Misconceptions

Clinicians play an integral role in addressing common misunderstandings around TD. Many families present with concerns around tics becoming worse, the safety of treatment, or worries that tics may return after successfully completing CBIT. Detailed below are common concerns and related suggestions for providing psychoeducation to address the concerns.

Tic worsening as a result of talking or thinking about them

While it is known that the frequency and severity of tics may increase when attention is placed on them, tic frequency/severity typically returns to baseline after shifting topics or distracting oneself with a different activity (Capriotti et al., 2014; Scotti-Degnan and Ford, 2020). Families should be given adequate psychoeducation around the role of social reinforcement in the cycle of tic expression throughout treatment. As contextual details are gathered throughout the course of CBIT, families should be reminded of how certain situations, patient demands, and affective states may lead tics to worsen. The functional intervention component of CBIT is designed expressly to address this issue.

Safety and tolerability

Both of the original CBIT RCTs found that participants receiving CBIT were no more likely than participants receiving supportive therapy and education to experience adverse events or to drop from the study. Despite initial concerns by some that awareness training and competing response would lead to increased anxiety, distractability, fatigue, and tic worsening, the rates of these events did not differ across the CBIT and PST conditions in the youth or adult studies (Piacentini et al., 2010; Wilhelm et al., 2012).

Rebound effects and symptom substitution

Likely a remnant of the early psychoanalytic conceptualization of tics as stemming from “psychogenic conflict” (Kushner, 1999), the common belief throughout the 20th century was that any attempt to suppress or otherwise block tic expression would lead to symptom substitution (other tics or psychiatric symptoms) or rebound effects (significant increase in tics beyond baseline levels). Not surprisingly, these and similar beliefs led the professional community to argue against behavioral interventions or any attempts to suppress or manage tics due to the fear of worsening tics or precipitating other psychiatric symptomatology. The positive outcomes from CBIT and other behavioral interventions have largely served to quell these concerns (Scahill et al., 2013). Additional research directly testing the link between tic suppression and rebound has also found no support for these myths (Capriotti et al., 2014; Himle et al., 2006). Rather, CBIT and other behavioral interventions have been shown to decrease targeted tics and support the use of strategies to decrease the frequency and severity of other tic symptoms (Capriotti et al., 2014; Franklin et al., 2010; Himle et al., 2006; McGuire et al., 2015).

Total tic remission with treatment

It is not uncommon for patients and families to enter treatment with the unrealistic expectation that treatment will be curative and lead to complete cessation of tics. Unfortunately, complete and durable remission of tics is rare across all treatment modalities. While CBIT is an effective approach to reducing tic severity within the context of an RCT, there is no evidence to suggest that most people will experience symptom remission as a result of CBIT (McGuire et al., 2015). It is imperative that clinicians work to address expectations for treatment early on with families.

Other Behavioral Treatments for Tics

All behavioral therapies for tics, including CBIT, seek to interrupt the stimulus–response pathways (urge leads to tic expression, which leads to relief from urge) that have been reinforced over time. However, the mechanism by which this is accomplished is as varied as the clinical utility and efficacy of the approaches.

Exposure plus response prevention

Exposure and response prevention (ERP) is an effective intervention for reducing tic severity in children and adults (Van De Griendt et al., 2018; Verdellen et al., 2011). The mechanism of action in ERP differs from CBIT/HRT, and relies upon habituation models of learning (Verdellen et al., 2011). In ERP, a patient increases the tolerance of interoceptive premonitory urges to tic across a variety of contexts (i.e., in vivo or imagined) while simultaneously suppressing tic expression for increasingly longer periods of time (Verdellen et al., 2011). The use of competing responses is not emphasized in ERP and all tics are targeted simultaneously, rather than systematically approached such as in CBIT.

In an RCT that compared ERP with HRT, Verdellen and colleagues (2004) found comparable reductions in tics across groups, although results were underpowered due to differences in therapeutic contact time between the ERP and HRT groups. More recently, Hollis and colleagues (2021) were able to demonstrate the utility of internet-delivered, parent-assisted, and therapist-supported ERP in children and adolescents. These researchers studied ERP specifically, theorizing that the absence of the competing response component of treatment would allow for easier online delivery with less therapeutic contact. Hollis et al. (2021) reported that those receiving online ERP were twice as likely to show a positive treatment response than those who received psychoeducation only, as evidenced by clinically significant reductions in YGTSS total tic scores from baseline to 3 months (d = –0.31), with a slightly increased effect at 6 months (d = –0.36). While the magnitude of effect was found to be approximately half of that in the face-to-face youth CBIT trial (Piacentini et al., 2010), Hollis and colleagues suggested that a direct comparison of the two studies’ effect sizes may be misleading due to the differences in implementation and the overall characteristics of the sample (e.g., higher baseline tic severity, higher rates of comorbidity, and less concomitant use of tic-suppressing medications). When balancing the smaller observed treatment effect size in the study by Hollis and colleagues with the greater convenience and reach of remote delivery, ERP can be considered an effective treatment for pediatric tics, especially for patients without access to more intensive and/or therapist-delivered care (Pringsheim and Piacentini, 2022).

Third-wave interventions

Elements of third-wave interventions have been incorporated into behavioral treatments for TD, which include acceptance-based approaches, mindfulness practices, and metacognitive training (Andrén et al., 2022; McGuire et al., 2018). Such strategies have been examined in the literature as adjuncts to HRT or as stand-alone treatments for tics (Andrén et al., 2022; McGuire et al., 2020a). Efforts to evaluate the feasibility and utility of these approaches have yielded modest results overall, indicating the need for further controlled examination. For example, Franklin and colleagues (2011) found that among a small sample of 13 adolescents with TD, those who received HRT plus components of Acceptance and Commitment Therapy demonstrated comparable reductions in tic severity and impairment as those receiving HRT alone. Although potentially promising, more work is needed to fully establish the utility of third-wave augmentation strategies. Other commonly used behavioral treatment components, including psychoeducation (Nussey et al., 2013), relaxation training (Bergin et al., 1998; Peterson and Azrin, 1992), and cognitive therapy (O’Connor et al., 2016), have not demonstrated efficacy for reducing tic severity when used as monotherapies.

CBIT Dissemination

In recent years, organizational, institutional, and technological efforts have been made to increase access to information around tics, provide recommendations for treatment, and assist in treatment delivery.

TicHelper

Modeled on the CBIT protocol, TicHelper (www.tichelper.com) is an 8-week, interactive online treatment program for youth ages 8 years through late adolescence with TD and their parents. TicHelper modules present CBIT material through a series of interactive activities, informational videos, and self-report assessments (Conelea and Wellen, 2017). Developed by Drs. Suzanne Mouton-Odum, Douglas Woods, and Michael Himle, with support from NIMH, TicHelper aims to enhance the accessibility of empirically based tic treatment for youth and their families. While unpublished data support preliminary feasibility, acceptability, and efficacy of the program, the dissemination potential, navigability, cost effectiveness, and evidence-based content support the potential of TicHelper as a valuable treatment resource.

TicTrainer

TicTrainer (www.tictrainer.com) is a free, web-based tool that uses a modified reward—enhanced version of ERP (RE-ERP), to address tics. This program is designed to increase the amount of time that the individual with TD is able to suppress tics through differential reinforcement of tic-free periods (Black and Black, 2018). TicTrainer requires both the individual patient (the “user”) and an external tic monitor (the “trainer”), such as a parent or therapist, to participate in training sessions. The trainer observes the user via webcam and clicks a button, upon seeing the user tic, which sends a signal to the user’s screen. The user earns more points/rewards for longer and longer periods of tic suppression. Notably, TicTrainer focuses exclusively on tic suppression and does not explicitly target the premonitory urge, which may impact the durability of gains over time. Similar to TicHelper, outcome research on the platform is yet to be published.

TAA-Behavior Therapy Institutes (TAA-BTI)

The Tourette Association of America sponsors several formal CBIT trainings each year (www.tourette.org/research-medical/cbit-for-practitioners ). Open to licensed clinicians, these two-day trainings cover the phenomenology and assessment of TD, the theory and science underlying CBIT, and a review and role-play of all CBIT components. As part of the training, attendees meet with Institute faculty in small groups to conceptualize and plan implementation of CBIT with active cases and then participate in three monthly consultation calls with their faculty to review delivery of the treatment, problem-solve any treatment complications, and plan subsequent treatment sessions.

Conclusion

For close to a century, behavioral interventions for managing tics associated with Tourette and other TDs were incorrectly considered ineffective and dangerous by the professional community, due, in large part, to unfounded fears that efforts to suppress tics would lead to a host of negative psychological, and even physical, outcomes (e.g., symptom substitution, tic rebound). Although pharmacotherapy remains the most common active intervention for Tourette and other TDs, the past two decades have seen remarkable growth in the acceptance and availability of behavioral interventions for tics, perhaps most notably, the CBIT, which has been designated as the first-line treatment option when available (Murphy et al., 2013; Pringsheim et al., 2019a; Verdellen et al., 2011). This growth has been spurred by a greater understanding of the quasi-volitional nature of some tics and the relationship between tic expression/suppression and a host of environmental factors, both internal (e.g., premonitory urges, anxiety) and external (e.g., settings, activities, responses, and consequences), that serve to trigger and maintain tics (Woods et al., 2008).

However, and despite the positive acute outcomes and long-term durability associated with CBIT, a significant proportion of participants in the original child trial (Piacentini et al., 2010) did not meet the response criteria following completion of the eight-session trial. While clinical use of CBIT, unconstrained by the rigid dosing characteristic of controlled treatment research, allows for more sessions and longer treatment as needed to achieve positive responses, additional research is needed to better understand the behavioral and neurobiological mechanisms underlying this treatment (e.g., Conelea et al., 2018). Research is also needed to optimize strategies for combining CBIT with pharmacologic, neuromodulatory, and other intervention modalities. Finally, additional work is necessary to foster larger scale dissemination of this treatment through remote telehealth delivery, app-based approaches, and other modalities that may arise.

Authors’ Contributions

K.K.: Conceptualization, investigation, and writing—original and review and editing; N.R.: Conceptualization, investigation, and writing—original and review and editing; J.P.: Conceptualization, investigation, writing—original and review and editing, resources, and supervision.

Footnotes

Author Disclosure Statement

Ms. Kohler and Rosen have no disclosures to report. Dr. Piacentini reports the following disclosures: (1) Research support from NIMH R01MH128595 and PCORI 19–08013; (2) paid advisor and equity share from Lumate Health, co-owner/founder of VirtuallyBetter Health, LLC; (3) publication royalties from Oxford University Press, Guilford Press, and Elsevier; and (4) travel and speaking honoraria from the International OCD Foundation and Tourette Association of America.

Funding Information

No funding was received for this article.

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