A novel, web-delivered, therapist-assisted version of exposure and response prevention therapy (ERP) may improve tic severity in Tourette syndrome (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 therapist-assisted internet-based ERP for tics or therapist-assisted internet education for tics for 10 weeks.
Results showed that both groups had significant improvement in tics from baseline to 3-month follow-up, but nearly half of the participants in the ERP group versus about a third of participants in the education group were responders classified the treatment.
“Therapist-assisted, web-delivered ERP and education were both associated with significant and clinically meaningful improvements in tic severity, although treatment response rates and satisfaction were significantly higher in the ERP group,” write the researchers, led by Per Andrén, PhD, Karolinska Institutet, Department of Clinical Neuroscience, Research Center for Child and Adolescent Psychiatry, Stockholm, Sweden.
“Implementing ERP digital intervention into mainstream healthcare would increase the availability of treatments for young people with TS or CTD,” they add.
The results were published online on August 15 JAMA network open.
Cognitive behavioral therapy is recommended by clinical guidelines as the first-line treatment for TS and CTD, but its availability is “very limited,” the researchers note. Therefore, “various formats of long-distance transmission have been proposed to improve access,” they write.
In a previous study, researchers developed an online-delivered behavioral therapy program for TS and CTD and found that ERP was “particularly well-suited to online guided exercise.”
These “promising results” spurred two parallel RCTs in England and Sweden that compared therapist-assisted web-delivered ERP to the “robust comparator model” of web-delivered education. The results of the UK ORBIT (Online Remote Behavioral Intervention for Tics) study showed that ERP was superior to the comparator in reducing tic severity. 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 comparison group (n=111 and 110, respectively). All were assessed at baseline, then at 3 and 5 weeks post-treatment, post-treatment, and 3 months post, which was the primary endpoint.
Most participants (91.4%) had TS, and 38% had one or more comorbid diagnoses—specifically, attention-deficit/hyperactivity disorder (15.4%) and anxiety disorders (14%). The majority of participants (85.5%) were not taking any medication at baseline.
The interventions consisted of 10 chapters, each completed weekly. Completion of treatment was defined a priori as completion of the first four children’s chapters, which contained “the core components of any intervention,” the researchers note.
In both interventions, children and parents were assisted by a licensed behavioral therapist, whose role was to “provide feedback, answer questions, and encourage adherence to treatment.”
The intervention focused on practicing tic suppression (response prevention) and gradually provoking premonitory urges, or the unpleasant sensations that typically precede tics. The latter should “make tic suppression more challenging,” the investigators write. The active comparison group consisted of education about TS and CTD and common comorbidities and behavioral exercises.
Tic severity, which was the primary outcome, was measured using the Yale Global Tic Severity Scale Total Tic Severity Score (YGTSS-TTSS).
Increased treatment access
From baseline to 3-month follow-up, there was a significant improvement in tic severity in both groups. However, there was a greater mean reduction in YGTSS-TTSS in the ERP versus comparison group (6.08 versus 5.29, respectively).
The mean YGTSS-TTSS score for the ERP group was 22.25 at baseline versus 16.17 at follow-up. For the comparison group, the values were 23.01 and 17.72, respectively.
The researchers report intention-to-treat analyzes showing that both groups “improved similarly over time” (interaction effect, -0.53; 95% CI, -1.28 to 0.22; P = 0.17).
However, at the 3-month follow-up, significantly more patients in the ERP group than in the comparator group were classified as responders to treatment (47.2% vs. 28.7%; odds ratio: 2.22; 95% CI: 1.27-3.90; P= 0.005).
Although both groups improved from baseline to 3-month follow-up on most secondary outcomes, including YGTSS impairment score, quality of life measures, obsessive-compulsive symptoms, and mood and feelings, only the ERP group showed improvements in clinical Comparison of Global Impression Severity and Improvement Scales and the parent-reported KIDSCREEN-10.
The mean intervention costs (time for therapist support) were “slightly higher” for the ERP than for the comparison group (mean difference $15.14; 95% CI $5.08 to $25.20), the investigators report . “ERP resulted in more patients responding to treatment at little additional cost compared to structured education,” they write.
They list several strengths of the study, including the use of an active comparator, nationwide recruitment, a large sample size, and very little data loss.
The limitations mentioned include the lack of a third group on the waiting list to control the natural passage of time; inclusion of a “generally mild group of participants”; and excluding participants with comorbid autism, potentially limiting the generalizability of the results.
Despite these limitations, “the results suggest that both web-delivered interventions could be implemented into mainstream healthcare to improve treatment access for children and adolescents with TS or CTD,” the researchers write.
They prefer the implementation of ERP to the educational intervention “because of the higher treatment response rates, likely cost-effectiveness, superior working alliance and satisfaction ratings, and the results of the parallel ORBIT study”.
Comment for Medscape Medical News Michael Okun, MD, director of the Norman Fixel Institute for Neurological Diseases, University of Florida Health, Gainesville, said the study “reinforces the idea that using telemedicine to bring therapies for tic disorders into the home is an important element for making will be more practical and available interventions.”
Okun, who was not involved in the research, added that the intervention is not only beneficial for adolescents but may also be beneficial for adults.
Although untested by current researchers, he noted that cognitive-behavioral intervention for tics is another therapy for the disorder that previous studies have shown to be effective when administered via telemedicine.
“Tic disorder therapies are challenging when multiple sessions over short periods of time are a requirement for success,” Okun said. “The use of telemedicine has opened a crucial 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, The Semel Institute for Neuroscience and Human Behavior, University of California, Los Angeles, notes that the intervention “has the potential to address several of the many significant barriers” patients and their families often face.
“The ability [for patients with TS] The use of a therapist-assisted remote delivery system could significantly improve both acceptance and capacity for caregiving and is a significant advance in the ability to offer therapeutic interventions in our field,” they write.
The study was funded by the Swedish Research Council on Health, Working Life and Welfare and the Swedish Research Council. The information provided by the investigators is in the original paper. Okun does not report any relevant financial relationships. Pringsheim reports that he has received research grants from Alberta Health and the Alberta Children’s Hospital Research Institute, and has been employed as a consultant on evidence-based medicine methods for the American Academy of Neurology. Piacentini reports receiving research support from NIMH, the Patient-Centered Outcomes Research Institute, the TLC Foundation for BFRBs, and the Nicholas Endowment; Spinnaker Health consultant fees; Publishing royalties from Guilford Press, Oxford University Press and Elsevier; and travel/speaker honoraria from the Tourette Association of America, the International OCD Foundation, and the TLC Foundation for BFRBs.
JAMA network open.Published online August 15, 2022. Full article, editorial
Batya Swift Yasgur, MA, LSW is a freelance writer with a consulting practice in Teaneck, New Jersey. She is a regular contributor to numerous medical publications, including Medscape and WebMD, and is the author of several consumer-focused health books, as well as Behind the Burqa: Our Lives in Afghanistan and How We Escaped to Freedom (the memoirs of two brave Afghan sisters who shared their stories with her). .
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