Online Remote Behavioural Intervention for Tics in 9 to 17-year-olds: the ORBIT RCT with embedded process and economic evaluation

Hollis, Chris; Hall, Charlotte L; Khan, Kareem; Le Novere, Marie; Marston, Louise; Jones, Rebecca; Hunter, Rachael; Brown, Beverley J; Sanderson, Charlotte; Andrén, Per; Bennett, Sophie D; Chamberlain, Liam R; Bethan Davies, E; Evans, Amber; Kouzoupi, Natalia; McKenzie, Caitlin; Heyman, Isobel; Kilgariff, Joseph; Glazebrook, Cristine; Mataix-Cols, David; Serlachius, Eva; Murray, Elizabeth; Murphy, Tara

Abstract

Background:

Behavioural therapy for tics is difficult to access, and little is known about its effectiveness when delivered online.

Objective:

To investigate the clinical and cost-effectiveness of an online-delivered, therapist and parent-supported therapy for young people with tic disorders.

Design:

Single-blind, parallel group, randomised controlled trial, with 3-month (primary end point) and 6-month post-randomisation follow-up. Participants were individually randomised (1:1), using on online system, with block randomisations, stratified by site. Naturalistic follow-up was conducted at 12- and 18-months post-randomisation when participants were free to access non-trial interventions. A subset of participants participated in a process evaluation.

Setting: Two hospitals (London and Nottingham) in England also accepting referrals from patient identification centres and online self-referrals.

Participants:

Children aged 9–17 years, with i) Tourette syndrome or chronic tic disorder; ii) a Yale Global Tic Severity Scale Total Tic Severity Score (YGTSS-TTSS) of 15 or more (or >10 with only motor or vocal tics); and iii) not received behavioural therapy for tics in the past 12 months or started/stopped medication for tics within the past 2 months.
Interventions: Either 10 weeks of online, remotely delivered, therapist-supported Exposure and Response Prevention therapy (intervention group) or online psychoeducation (control).

Outcome:

Primary outcome: YGTSS-TTSS 3 months post-randomisation; analysis done in all randomised patients for whom data were available. Secondary outcomes included low mood, anxiety, treatment satisfaction and health resource use. Quality-of-life-adjusted-years are derived from parent completed quality-of-life measures. All trial staff, statisticians and the chief investigator were masked to group allocation.

Results:

224 participants were randomised to the intervention (n=112) or control (n=112). Participants were mostly male (n=177; 79%) with a mean age of 12 years. At 3 months the estimated mean difference in YGTSS-TTSS between the groups adjusted for baseline and site was –2.29 points (95% CI –3.86 to –0.71) in favour of therapy (effect size –0.31; 95% CI –0.52 to –0.10). This effect was sustained throughout to the final follow-up at 18 months -2.01 points (95% CI: -3.86 to -0.15, effect size -0.27; 95%CI -0.52 to -0.02). At 18 months the mean incremental cost per participant of the intervention compared to the control was £662 (95% CI -£59 to £1384) with a mean incremental quality-adjusted-life-year of 0.040 (95% CI -0.004 to 0.083) per participant. The mean incremental cost per quality-adjusted-life-year gained was £16,708.

The intervention was acceptable and delivered with high fidelity. Parental engagement predicted child engagement and more positive clinical outcomes.

Harms:

Two serious, unrelated, adverse events occurred in the control group.
Limitations: We cannot separate the effects of digital online delivery and the therapy itself. The sample was predominately white, British, limiting generalisability. The design did not compare to face-to-face services. Future work: Future work should compare online to face-to-face therapy and explore how to embed the intervention in clinical practice.

Conclusion:

Online, therapist supported behavioural therapy for young people with tic disorders is clinically- and cost-effective in reducing tics with durable benefits extending up to 18 months.

Study registration: ISRCTN (ISRCTN70758207); ClinicalTrials.gov (NCT03483493). The trial is now complete.

Citation

Hollis, C., Hall, C. L., Khan, K., Le Novere, M., Marston, L., Jones, R., Hunter, R., Brown, B. J., Sanderson, C., Andrén, P., Bennett, S. D., Chamberlain, L. R., Bethan Davies, E., Evans, A., Kouzoupi, N., McKenzie, C., Heyman, I., Kilgariff, J., Glazebrook, C., Mataix-Cols, D., …Murphy, T. (2023). Online Remote Behavioural Intervention for Tics in 9 to 17-year-olds: the ORBIT RCT with embedded process and economic evaluation. Health Technology Assessment, 27(18), 1-120. https://doi.org/10.3310/cpms3211

• Journal Article Type: Article
• Acceptance Date: Dec 21, 2022
• Publication Date: Oct 25, 2023
• Deposit Date: Jan 6, 2023
• Publicly Available Date: Oct 25, 2023
• Journal: Health Technology Assessment
• Print ISSN: 1366-5278
• Electronic ISSN: 2046-4924
• Publisher: NIHR Journals Library
• Peer Reviewed: Peer Reviewed
• Volume: 27
• Issue: 18
• Pages: 1-120
• DOI: https://doi.org/10.3310/cpms3211
• Public URL: https://nottingham-repository.worktribe.com/output/15717872
• Publisher URL: https://www.journalslibrary.nihr.ac.uk/HTA/#/
• Additional Information: Free to read: This content has been made freely available to all.; contractual_start_date: 10-2017; editorial review begun: 07-2022; Accepted for publication: 12-2022