Clinical Context

Tourette syndrome (TS) is a common and impactful health condition, affecting approximately 1% of school age children. Although medications such as alpha-2 agonists or dopamine antagonists can be used to treat TS, behavioral therapy is a preferred option for many children. An editorial by Pringsheim and Placentini, which accompanies the current study, describes the practice of comprehensive behavioral intervention for tics (CBIT).

Tics are typically a negative response to unpleasant stimuli among children. CBIT does not deny noxious stimuli to children but instead teaches techniques to suppress tics in the setting of such stimuli. Moreover, the typical course of CBIT involves exposing patients to increasing levels of noxious stimuli to build their strength in tic suppression. This treatment modality has been demonstrated to improve tic symptoms over the long-term after completion of therapy.

Could a form of CBIT be effectively delivered online? The current study addresses this question.

Study Synopsis and Perspective

A novel internet-delivered, therapist-supported version of exposure and response prevention (ERP) therapy can yield improvements in tic severity in TS or chronic tic disorder (CTD), new research suggests.

In a randomized controlled trial (RCT), more than 200 adolescents with TS or CTD received either 10 weeks of therapist-supported internet-delivered ERP for tics or therapist-supported internet education for tics.

Results showed that both groups had significant improvement in tics from baseline to 3-month follow-up, but almost half of participants in the ERP group vs about one third of participants in the education group were classified as treatment responders.

"Therapist-supported internet-delivered ERP and education were both associated with significantly and clinically meaningful improvements in tic severity, although treatment response rates and satisfaction were significantly higher in the ERP group," write the investigators, led by Per Andrén, PhD, Karolinska Institutet, Department of Clinical Neuroscience, Child and Adolescent Psychiatry Research Center, Stockholm, Sweden.

"Implementation of the digital ERP intervention into regular health care would increase availability of treatment for young people with TS or CTD," they add.

The findings were published online August 15 in JAMA Network Open.^[1]

Behavior therapy is recommended by clinical guidelines as first-line treatment for TS and CTD, but its availability is "very limited," the researchers note. Therefore, "various formats of remote delivery have been proposed to improve access," they write.

In a previous study, the investigators developed an internet-delivered behavior therapy program for TS and CTD and found that ERP was "particularly well-suited to guided online delivery."

These "promising results" spurred 2 parallel RCTs in England and Sweden that compared therapist-supported internet-delivered ERP with the "robust comparator" of internet-delivered education. Results from the British Online Remote Behavioral Intervention for Tics (ORBIT) trial showed that ERP was superior to the comparator in reducing tic severity.^[3] The current analysis presents the results of the Swedish RCT.

Participants (n=221; 68.8% boys; mean age, 12.1 years) were randomly assigned to either the ERP or comparator groups (n=111 and 110, respectively). All were assessed at baseline and then at 3 and 5 weeks into treatment, at posttreatment, and 3 months afterward, which constituted the primary endpoint.

Most of the participants (91.4%) had TS, and 38.0% had 1 or more comorbid diagnosis, particularly attention-deficit/hyperactivity disorder (15.4%) and anxiety disorders (14.0%). The majority of participants (85.5%) were not receiving medications at baseline.

The interventions consisted of 10 chapters, each completed weekly. Treatment completion was defined a priori as the completion of the first four child chapters, which contained "the core ingredients of each intervention," the researchers note.

In both interventions, children and parents were supported by a designated therapist trained in behavior therapy, whose role was to "provide feedback, answer questions, and encourage treatment adherence."

The intervention focused on practicing tic suppression (response prevention) and gradually provoking premonitory urges, or the unpleasant sensations typically preceding tics. The latter was designed to "make the tic suppression more challenging," the investigators write. The active comparator consisted of education about TS and CTD and common comorbidities, as well as about behavioral exercises.

Tic severity, which was the primary outcome, was measured by the Total Tic Severity Score of the Yale Global Tic Severity Scale (YGTSS-TTSS).

From baseline to 3-month follow-up, there was significant improvement in tic severity in both groups. However, there was a higher mean reduction on the YGTSS-TTSS in the ERP vs the comparator group (6.08 vs 5.29, respectively).

The mean YGTSS-TTSS score for the ERP group at baseline was 22.25 vs 16.17 at follow-up. For the comparator group, the scores were 23.01 vs 17.72, respectively.

The investigators report intention-to-treat analyses showing that both groups "improved similarly over time" (interaction effect, −0.53; 95% confidence interval [CI], −1.28 to 0.22; P=.17).

However, at 3-month follow-up, significantly more patients were classified as treatment responders in the ERP group vs the comparator group (47.2% vs 28.7%, respectively; odds ratio, 2.22; 95% CI, 1.27-3.90; P=.005).

Although both groups improved from baseline to 3-month follow-up on most secondary outcomes, including the YGTSS Impairment Score, quality-of-life measures, obsessive-compulsive symptoms, and mood and feelings, only the ERP group showed improvements on the Clinical Global Impression Severity and Improvement Scales and the parent-reported KIDSCREEN-10.

The mean intervention costs (therapist-support time) were "slightly higher" for the ERP vs the comparator group (mean difference, $15.14; 95% CI, $5.08-$25.20), the investigators report. "ERP resulted in more treatment responders at little additional cost compared with structured education," they write.

The researchers list several strengths of the study, including use of an active comparator, nationwide recruitment, a large sample size, and very low data attrition.

Limitations cited include the absence of a third wait-listed group to control for the natural passage of time, inclusion of a "generally mild group of participants," and exclusion of participants with comorbid autism, potentially limiting the generalizability of the findings.

Despite these limitations, the findings "suggest that both internet-delivered interventions could be implemented into regular health care to increase treatment access for children and adolescents with TS or CTD," the researchers write.

They favor the implementation of ERP vs the educational intervention "based on its higher treatment response rates, likely cost-effectiveness, superior working alliance and satisfaction ratings, as well as the results from the parallel ORBIT trial."

Commenting for Medscape Medical News, Michael Okun, MD, director of the Norman Fixel Institute for Neurological Diseases, University of Florida Health, Gainesville, said that the study "reinforces the idea that using telemedicine to bring therapies for tic disorders into the home will be an important element to making interventions more practical and available."

Dr Okun, who was not involved with the research, added that the intervention has utility not only for youth but also potentially for adults.

Although not tested by the current investigators, he noted that cognitive-behavioral intervention for tics is another therapy used for the disorder that has been shown, in previous studies, to be effective when delivered via telemedicine.

"Therapies for tic disorders are challenging to deliver when multiple sessions over short periods of time are a requirement for success," Dr Okun said. "The use of telemedicine has opened a critical door to the future."

In an accompanying editorial, Tamara Pringsheim, MD, Cumming School of Medicine, Department of Clinical Neuroscience, Psychiatry, Pediatrics, and Community Health Sciences, University of Calgary, Alberta, Canada, and John Piacentini, PhD, Department of Psychiatry and Biobehavioral Sciences, Semel Institute for Neuroscience and Human Behavior, University of California, Los Angeles, note that the intervention "has the potential to address several of the many significant barriers" often faced by patients and their families.^[2]

"The ability [for patients with TS] to use a remote delivery system with therapist support could greatly increase both acceptability and capacity for care and is a meaningful advance in the ability to provide therapeutic interventions in our field," they write.

The study was funded by the Swedish Research Council for Health, Working Life and Welfare, and the Swedish Research Council. The investigators' disclosures are in the original paper. Dr Okun reports no relevant financial relationships. Dr Pringsheim reports having received research funding from Alberta Health and the Alberta Children's Hospital Research Institute and employment as an evidence-based medicine methodology consultant for the American Academy of Neurology. Dr Piacentini reports receiving research support from the National Institute of Mental Health, the Patient-Centered Outcomes Research Institute, the TLC Foundation for BFRBs, and the Nicholas Endowment; consultant fees from Spinnaker Health; publication royalties from Guilford Press, Oxford University Press, and Elsevier; and travel/speaking honoraria from the Tourette Association of America, International OCD Foundation, and TLC Foundation for BFRBs.

JAMA Netw Open. Published online August 15, 2022.

Clinical Implications

• CBIT promotes active tic suppression among patients with TS while exposing them to increasing levels of noxious stimuli.
• The current study demonstrates that both ERP and disease education can improve the frequency of tics among children and adolescents, whether measured by clinicians, patients, or parents. Satisfaction with treatment was higher in the ERP vs education groups.

Earn Credit