Therapist-Supported Internet-Delivered Exposure and Response Prevention for Children and Adolescents with Tourette Syndrome: A Randomized Clinical Trial

Andrén, Per; Holmsved, Moa; Ringberg, Helene; Wachtmeister, Vera; Isomura, Kayoko; Aspvall, Kristina; Lenhard, Fabian; Hall, Charlotte L.; Davies, Bethan; Murphy, Tara; Hollis, Chris; Sampaio, Filipa; Feldman, Inna; Bottai, Matteo; Serlachius, Eva; Andersson, Erik; Fernández De La Cruz, Lorena; Mataix-Cols, David

Abstract

Importance: The availability of behavior therapy for individuals with Tourette syndrome (TS) and chronic tic disorder (CTD) is limited. Objective: To determine the efficacy and cost-effectiveness of internet-delivered exposure and response prevention (ERP) for children and adolescents with TS or CTD. Design, Setting, and Participants: This single-masked, parallel group, superiority randomized clinical trial with nationwide recruitment was conducted at a research clinic in Stockholm, Sweden. Out of 615 individuals assessed for eligibility, 221 participants meeting diagnostic criteria for TS or CTD and aged 9 to 17 years were included in the study. Enrollment began in April 2019 and ended in April 2021. Data were analyzed between October 2021 and March 2022. Interventions: Participants were randomized to 10 weeks of therapist-supported internet-delivered ERP for tics (111 participants) or to therapist-supported internet-delivered education for tics (comparator group, 110 participants). Main Outcomes and Measures: The primary outcome was change in tic severity from baseline to the 3-month follow-up as measured by the Total Tic Severity Score of the Yale Global Tic Severity Scale (YGTSS-TTSS). YGTSS-TTSS assessors were masked to treatment allocation. Treatment response was operationalized as a score of 1 ("Very much improved") or 2 ("Much improved") on the Clinical Global Impression-Improvement scale. Results: Data loss was minimal, with 216 of 221 participants (97.7%) providing primary outcome data. Among randomized participants (152 [68.8%] boys; mean [SD] age, 12.1 [2.3] years), tic severity improved significantly, with a mean reduction of 6.08 points on the YGTSS-TTSS in the ERP group (mean [SD] at baseline, 22.25 [5.60]; at 3-month follow-up, 16.17 [6.82]) and 5.29 in the comparator (mean [SD] at baseline, 23.01 [5.92]; at 3-month follow-up, 17.72 [7.11]). Intention-to-treat analyses showed that the 2 groups improved similarly over time (interaction effect, -0.53; 95% CI, -1.28 to 0.22; P =.17). Significantly more participants were classified as treatment responders in the ERP group (51 of 108 [47.2%]) than in the comparator group (31 of 108 [28.7%]) at the 3-month follow-up (odds ratio, 2.22; 95% CI, 1.27 to 3.90). ERP resulted in more treatment responders at little additional cost compared with structured education. The incremental cost per quality-adjusted life-year gained was below the Swedish willingness-to-pay threshold, at which ERP had a 66% to 76% probability of being cost-effective. Conclusions and Relevance: Both interventions were associated with clinically meaningful improvements in tic severity, but ERP led to higher response rates at little additional cost. Trial Registration: ClinicalTrials.gov identifier: NCT03916055.

Citation

Andrén, P., Holmsved, M., Ringberg, H., Wachtmeister, V., Isomura, K., Aspvall, K., …Mataix-Cols, D. (2022). Therapist-Supported Internet-Delivered Exposure and Response Prevention for Children and Adolescents with Tourette Syndrome: A Randomized Clinical Trial. JAMA Network Open, 5(8), Article e2225614. https://doi.org/10.1001/jamanetworkopen.2022.25614

• Journal Article Type: Article
• Acceptance Date: Jun 22, 2022
• Online Publication Date: Aug 15, 2022
• Publication Date: Aug 15, 2022
• Deposit Date: Jun 22, 2022
• Publicly Available Date: Aug 17, 2022
• Journal: JAMA Network Open
• Electronic ISSN: 2574-3805
• Publisher: American Medical Association
• Peer Reviewed: Peer Reviewed
• Volume: 5
• Issue: 8
• Article Number: e2225614
• DOI: https://doi.org/10.1001/jamanetworkopen.2022.25614
• Keywords: General Medicine
• Public URL: https://nottingham-repository.worktribe.com/output/8632245
• Publisher URL: https://jamanetwork.com/journals/jamanetworkopen/fullarticle/2795148