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

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.

Conflict of interest statement

Conflict of Interest Disclosures: Dr Serlachius reported receiving grant support from Kavli Trust, Region Stockholm, Forte Swedish Research Council for Health, Working Life and Welfare, Frimurare Barnhuset Trust, Jane and Dan Olsson Foundation, and Riksbankens jubileumsfond outside the submitted work. Dr Andersson reported royalties from Natur & Kultur from a self-help book on health anxiety. Dr Fernández de la Cruz reported receiving grants from Swedish Research Council for Health, Working Life and Welfare (Forte), Region Stockholm (ALF), Hjärt-Lungfonden, Åke Wibergs Stiftelse, and Karolinska Institutet outside the submitted work; she reported receiving personal fees from UpToDate, Wolters Kluwer Health (contributing articles), and Elsevier (editorial work) outside the submitted work. Dr Mataix-Cols reported receiving personal fees from UptoDate Inc, Wolters Kluwer Health, and Elsevier outside the submitted work. No other disclosures were reported.

Figures

Figure 1.. Flow Diagram of Study Participation

BT indicates behavior therapy; CTD, chronic tic disorder; ERP, therapist-supported internet-delivered exposure with response prevention for children and adolescents with Tourette syndrome or chronic tic disorder; TS, Tourette syndrome; YGTSS-TTSS, Yale Global Tic Severity Scale–Total Tic Severity Score. aOther included families that did not want to participate in a research study, did not feel motivated, did not have the energy for assessment or treatment, did not have enough time, did not want to be video recorded, did not want a TS or CTD diagnosis to be recorded in their patient record, with parents who wanted to participate but not the child, as well as cases where no specific reasons were specified.

Figure 2.. Cost-effectiveness Planes With QALYs as the Outcome for 3 Costing Perspectives

All 3 cost-effectiveness planes compare ERP with therapist-supported internet-delivered education for children and adolescents with Tourette syndrome or chronic tic disorder (ie, the comparator) using QALYs as the outcome. In panel A, the health care organization perspective includes costs of the ERP or comparator interventions (ie, the therapist-support time). In panel B, the health care sector perspective includes costs of the ERP or comparator interventions, health care visits, and medication or supplements. In panel C, the societal perspective includes costs of the ERP or comparator interventions, health care visits, medication or supplements, and other sector costs (eg, productivity losses, child school absenteeism). ERP indicates therapist-supported internet-delivered exposure with response prevention for children and adolescents with Tourette syndrome or chronic tic disorder; QALY, quality-adjusted life-year.