Long‐term clinical and cost‐effectiveness of a therapist‐supported online remote behavioural intervention for tics in children and adolescents: extended 12‐ and 18‐month follow‐up of a single‐blind randomised controlled trial

Chris Hollis; Charlotte l. Hall; Kareem Khan; Rebecca Jones; Louise Marston; Marie Le novere; Rachael Hunter; Per Andrén; Sophie D. Bennett; Beverley J. Brown; Liam r. Chamberlain; E. Bethan Davies; Amber Evans; Natalia Kouzoupi; Caitlin Mckenzie; Charlotte Sanderson; Isobel Heyman; Joseph Kilgariff; Cristine Glazebrook; David Mataix‐cols; Eva Serlachius; Elizabeth Murray; Tara Murphy

doi:10.1111/jcpp.13756

Long‐term clinical and cost‐effectiveness of a therapist‐supported online remote behavioural intervention for tics in children and adolescents: extended 12‐ and 18‐month follow‐up of a single‐blind randomised controlled trial

Chris Hollis, Charlotte l. Hall, Kareem Khan, Rebecca Jones, Louise Marston, Marie Le novere, Rachael Hunter, Per Andrén, Sophie D. Bennett, Beverley J. Brown, Liam r. Chamberlain, E. Bethan Davies, Amber Evans, Natalia Kouzoupi, Caitlin Mckenzie, Charlotte Sanderson, Isobel Heyman, Joseph Kilgariff, Cristine Glazebrook, David Mataix‐colsEva Serlachius, Elizabeth Murray, Tara Murphy

Research output: Contribution to journal › Article › peer-review

6 Citations (Scopus)

Abstract

Background: Little is known about the long-term effectiveness of behavioural therapy for tics. We aimed to assess the long-term clinical and cost-effectiveness of online therapist-supported exposure and response prevention (ERP) therapy for tics 12 and 18 months after treatment initiation. Methods: ORBIT (online remote behavioural intervention for tics) was a two-arm (1:1 ratio), superiority, single-blind, multicentre randomised controlled trial comparing online ERP for tics with online psychoeducation. The trial was conducted across two Child and Adolescent Mental Health Services in England. Participants were recruited from these two sites, across other clinics in England, or by self-referral. This study was a naturalistic follow-up of participants at 12- and 18-month postrandomisation. Participants were permitted to use alternative treatments recommended by their clinician. The key outcome was the Yale Global Tic Severity Scale Total Tic Severity Score (YGTSS-TTSS). A full economic evaluation was conducted. Registrations are ISRCTN (ISRCTN70758207); ClinicalTrials.gov (NCT03483493). Results: Two hundred and twenty-four participants were enrolled: 112 to ERP and 112 to psychoeducation. The sample was predominately male (177; 79%) and of white ethnicity (195; 87%). The ERP intervention reduced baseline YGTSS-TTSS by 2.64 points (95% CI: −4.48 to −0.79) with an effect size of −0.36 (95% CI: −0.61 to −0.11) after 12 months and by 2.01 points (95% CI: −3.86 to −0.15) with an effect size of −0.27 (95% CI -0.52 to −0.02) after 18 months, compared with psychoeducation. Very few participants (<10%) started new tic treatment during follow-up. The cost difference in ERP compared with psychoeducation was £304.94 (−139.41 to 749.29). At 18 months, the cost per QALY gained was £16,708 for ERP compared with psychoeducation. Conclusions: Remotely delivered online ERP is a clinical and cost-effective intervention with durable benefits extending for up to 18 months. This represents an efficient public mental health approach to increase access to behavioural therapy and improve outcomes for tics.

Original language	English
Pages (from-to)	941-951
Number of pages	11
Journal	Journal of child psychology and psychiatry
Volume	64
Issue number	6
Early online date	17 Jan 2023
DOIs	https://doi.org/10.1111/jcpp.13756
Publication status	Published - Jun 2023

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Hollis, C., Hall, C. L., Khan, K., Jones, R., Marston, L., Le novere, M., Hunter, R., Andrén, P., Bennett, S. D., Brown, B. J., Chamberlain, L. R., Davies, E. B., Evans, A., Kouzoupi, N., Mckenzie, C., Sanderson, C., Heyman, I., Kilgariff, J., Glazebrook, C., ... Murphy, T. (2023). Long‐term clinical and cost‐effectiveness of a therapist‐supported online remote behavioural intervention for tics in children and adolescents: extended 12‐ and 18‐month follow‐up of a single‐blind randomised controlled trial. Journal of child psychology and psychiatry, 64(6), 941-951. https://doi.org/10.1111/jcpp.13756

Hollis, Chris ; Hall, Charlotte l. ; Khan, Kareem et al. / Long‐term clinical and cost‐effectiveness of a therapist‐supported online remote behavioural intervention for tics in children and adolescents : extended 12‐ and 18‐month follow‐up of a single‐blind randomised controlled trial. In: Journal of child psychology and psychiatry. 2023 ; Vol. 64, No. 6. pp. 941-951.

@article{50b68b412ac94986b730ecdc1f1b7249,

title = "Long‐term clinical and cost‐effectiveness of a therapist‐supported online remote behavioural intervention for tics in children and adolescents: extended 12‐ and 18‐month follow‐up of a single‐blind randomised controlled trial",

abstract = "Background: Little is known about the long-term effectiveness of behavioural therapy for tics. We aimed to assess the long-term clinical and cost-effectiveness of online therapist-supported exposure and response prevention (ERP) therapy for tics 12 and 18 months after treatment initiation. Methods: ORBIT (online remote behavioural intervention for tics) was a two-arm (1:1 ratio), superiority, single-blind, multicentre randomised controlled trial comparing online ERP for tics with online psychoeducation. The trial was conducted across two Child and Adolescent Mental Health Services in England. Participants were recruited from these two sites, across other clinics in England, or by self-referral. This study was a naturalistic follow-up of participants at 12- and 18-month postrandomisation. Participants were permitted to use alternative treatments recommended by their clinician. The key outcome was the Yale Global Tic Severity Scale Total Tic Severity Score (YGTSS-TTSS). A full economic evaluation was conducted. Registrations are ISRCTN (ISRCTN70758207); ClinicalTrials.gov (NCT03483493). Results: Two hundred and twenty-four participants were enrolled: 112 to ERP and 112 to psychoeducation. The sample was predominately male (177; 79%) and of white ethnicity (195; 87%). The ERP intervention reduced baseline YGTSS-TTSS by 2.64 points (95% CI: −4.48 to −0.79) with an effect size of −0.36 (95% CI: −0.61 to −0.11) after 12 months and by 2.01 points (95% CI: −3.86 to −0.15) with an effect size of −0.27 (95% CI -0.52 to −0.02) after 18 months, compared with psychoeducation. Very few participants (<10%) started new tic treatment during follow-up. The cost difference in ERP compared with psychoeducation was £304.94 (−139.41 to 749.29). At 18 months, the cost per QALY gained was £16,708 for ERP compared with psychoeducation. Conclusions: Remotely delivered online ERP is a clinical and cost-effective intervention with durable benefits extending for up to 18 months. This represents an efficient public mental health approach to increase access to behavioural therapy and improve outcomes for tics.",

author = "Chris Hollis and Hall, {Charlotte l.} and Kareem Khan and Rebecca Jones and Louise Marston and {Le novere}, Marie and Rachael Hunter and Per Andr{\'e}n and Bennett, {Sophie D.} and Brown, {Beverley J.} and Chamberlain, {Liam r.} and Davies, {E. Bethan} and Amber Evans and Natalia Kouzoupi and Caitlin Mckenzie and Charlotte Sanderson and Isobel Heyman and Joseph Kilgariff and Cristine Glazebrook and David Mataix‐cols and Eva Serlachius and Elizabeth Murray and Tara Murphy",

note = "Funding Information: This research was funded by the NIHR Heath Technology Assessment (HTA) (Ref 16/19/02). The views expressed are those of the author(s) and not necessarily those of the NHS, the NIHR or the Department of Health. C.H., C.L.H., E.B.D. and C.G. acknowledge the financial support of the NIHR Nottingham Biomedical Research Centre and NIHR MindTech MedTech Co‐operative. The study sponsor and funders have no role in study design, including collection, management, analysis and interpretation of data; writing of the report; and the decision to submit the report for publication. The corresponding author (C.H.) had full access to all the data in the study and had final responsibility for the decision to submit for publication. Funding Information: The authors would like to thank additional members who supported the ORBIT team at UCL and University of Nottingham. The authors would like to thank Tourettes Action for their support with the trial. The authors would like to thank their Patient and Public Involvement (PPI) members for their help. The authors thank members of their Trial Steering Committee, chaired by Prof Edmund Sonuga-Barke and the members of their Data Monitoring Committee Chaired by Prof Stephen Scott. The authors would also like to thank Angela Summerfield for her continued administrative support and help. The authors would also like to thank their patient identification centres (PIC) who recruited patients into the ORBIT study: Guys and St Thomas NHS Foundation Trust, Oxford Health NHS Foundation Trust, North East London NHS Foundation Trust, Pennine Care NHS Foundation Trust, Sussex Partnership NHS Foundation Trust, Central Manchester University Hospitals NHS Foundation Trust, Cumbria Partnership NHS Foundation Trust, Leicestershire Partnership NHS Trust, United Lincolnshire Hospitals NHS Trust, East Kent Hospitals University NHS Foundation Trust, Northern Lincolnshire and Goole NHS Foundation Trust, Northumberland Tyne and Wear NHS Foundation Trust, Nottingham University Hospitals NHS Trust, Birmingham Women's and Children's NHS Foundation Trust and Northamptonshire Healthcare NHS Foundation Trust. The authors also acknowledge that all research at Great Ormond Street Hospital NHS Foundation Trust and UCL Great Ormond Street Institute of Child Health is made possible by the NIHR Great Ormond Street Hospital Biomedical Research Centre. This work used services from the eHealth Core Facility at Karolinska Institutet, which is supported by the Strategic Research Area Healthcare Science (SFO-V). This research was funded by the NIHR Heath Technology Assessment (HTA) (Ref 16/19/02). The views expressed are those of the author(s) and not necessarily those of the NHS, the NIHR or the Department of Health. C.H., C.L.H., E.B.D. and C.G. acknowledge the financial support of the NIHR Nottingham Biomedical Research Centre and NIHR MindTech MedTech Co-operative. The study sponsor and funders have no role in study design, including collection, management, analysis and interpretation of data; writing of the report; and the decision to submit the report for publication. The corresponding author (C.H.) had full access to all the data in the study and had final responsibility for the decision to submit for publication. Aside from receiving funding from NIHR to support their salaries C.L.H., L.M., E.B.D., R.J., K.K., B.J.B., C.S., C.M., E.S., P.A., I.H., J.K., R.H., M.L.N., A.E., L.R.C., N.K., S.B., C.G. and E.M. declare no conflict of interest. C.H. declares he was the principal investigator on a grant from the National Institute of Health Research (NIHR) Health Technology Assessment programme to conduct an Evidence Synthesis on the treatments for tics and Tourette syndrome in children and young people{\textquoteright} HTA Project:10/142/01. D.M-C. reports personal fees from UpToDate Inc. outside the submitted work. T.M. reports personal fees from Royalties from Jessica Kingsley Publishers received for the following publications: The Tourettes Survival Kit: Tools for Young Adults with Tics (2019) by Tara Murphy and Damon Millar personal fees from Tic Disorders: A Guide for Parents and Professionals (2016) by Uttom Chowdhury and Tara Murphy from The Incredible Teenage Brain: Everything You Need to Know to Unlock Your Teen's Potential (2019) by Bettina Hohnen, Jane Gilmour & Tara Murphy, outside the submitted work. Funding Information: The authors also acknowledge that all research at . This work used services from the eHealth Core Facility at Karolinska Institutet, which is supported by the Strategic Research Area Healthcare Science (SFO‐V). Great Ormond Street Hospital NHS Foundation Trust and UCL Great Ormond Street Institute of Child Health is made possible by the NIHR Great Ormond Street Hospital Biomedical Research Centre Publisher Copyright: {\textcopyright} 2023 The Authors. Journal of Child Psychology and Psychiatry published by John Wiley & Sons Ltd on behalf of Association for Child and Adolescent Mental Health.",

year = "2023",

month = jun,

doi = "10.1111/jcpp.13756",

language = "English",

volume = "64",

pages = "941--951",

journal = "Journal of child psychology and psychiatry",

issn = "0021-9630",

number = "6",

}

Hollis, C, Hall, CL, Khan, K, Jones, R, Marston, L, Le novere, M, Hunter, R, Andrén, P, Bennett, SD, Brown, BJ, Chamberlain, LR, Davies, EB, Evans, A, Kouzoupi, N, Mckenzie, C, Sanderson, C, Heyman, I, Kilgariff, J, Glazebrook, C, Mataix‐cols, D, Serlachius, E, Murray, E & Murphy, T 2023, 'Long‐term clinical and cost‐effectiveness of a therapist‐supported online remote behavioural intervention for tics in children and adolescents: extended 12‐ and 18‐month follow‐up of a single‐blind randomised controlled trial', Journal of child psychology and psychiatry, vol. 64, no. 6, pp. 941-951. https://doi.org/10.1111/jcpp.13756

Long‐term clinical and cost‐effectiveness of a therapist‐supported online remote behavioural intervention for tics in children and adolescents: extended 12‐ and 18‐month follow‐up of a single‐blind randomised controlled trial. / Hollis, Chris; Hall, Charlotte l.; Khan, Kareem et al.
In: Journal of child psychology and psychiatry, Vol. 64, No. 6, 06.2023, p. 941-951.

Research output: Contribution to journal › Article › peer-review

TY - JOUR

T1 - Long‐term clinical and cost‐effectiveness of a therapist‐supported online remote behavioural intervention for tics in children and adolescents

T2 - extended 12‐ and 18‐month follow‐up of a single‐blind randomised controlled trial

AU - Hollis, Chris

AU - Hall, Charlotte l.

AU - Khan, Kareem

AU - Jones, Rebecca

AU - Marston, Louise

AU - Le novere, Marie

AU - Hunter, Rachael

AU - Andrén, Per

AU - Bennett, Sophie D.

AU - Brown, Beverley J.

AU - Chamberlain, Liam r.

AU - Davies, E. Bethan

AU - Evans, Amber

AU - Kouzoupi, Natalia

AU - Mckenzie, Caitlin

AU - Sanderson, Charlotte

AU - Heyman, Isobel

AU - Kilgariff, Joseph

AU - Glazebrook, Cristine

AU - Mataix‐cols, David

AU - Serlachius, Eva

AU - Murray, Elizabeth

AU - Murphy, Tara

N1 - Funding Information: This research was funded by the NIHR Heath Technology Assessment (HTA) (Ref 16/19/02). The views expressed are those of the author(s) and not necessarily those of the NHS, the NIHR or the Department of Health. C.H., C.L.H., E.B.D. and C.G. acknowledge the financial support of the NIHR Nottingham Biomedical Research Centre and NIHR MindTech MedTech Co‐operative. The study sponsor and funders have no role in study design, including collection, management, analysis and interpretation of data; writing of the report; and the decision to submit the report for publication. The corresponding author (C.H.) had full access to all the data in the study and had final responsibility for the decision to submit for publication. Funding Information: The authors would like to thank additional members who supported the ORBIT team at UCL and University of Nottingham. The authors would like to thank Tourettes Action for their support with the trial. The authors would like to thank their Patient and Public Involvement (PPI) members for their help. The authors thank members of their Trial Steering Committee, chaired by Prof Edmund Sonuga-Barke and the members of their Data Monitoring Committee Chaired by Prof Stephen Scott. The authors would also like to thank Angela Summerfield for her continued administrative support and help. The authors would also like to thank their patient identification centres (PIC) who recruited patients into the ORBIT study: Guys and St Thomas NHS Foundation Trust, Oxford Health NHS Foundation Trust, North East London NHS Foundation Trust, Pennine Care NHS Foundation Trust, Sussex Partnership NHS Foundation Trust, Central Manchester University Hospitals NHS Foundation Trust, Cumbria Partnership NHS Foundation Trust, Leicestershire Partnership NHS Trust, United Lincolnshire Hospitals NHS Trust, East Kent Hospitals University NHS Foundation Trust, Northern Lincolnshire and Goole NHS Foundation Trust, Northumberland Tyne and Wear NHS Foundation Trust, Nottingham University Hospitals NHS Trust, Birmingham Women's and Children's NHS Foundation Trust and Northamptonshire Healthcare NHS Foundation Trust. The authors also acknowledge that all research at Great Ormond Street Hospital NHS Foundation Trust and UCL Great Ormond Street Institute of Child Health is made possible by the NIHR Great Ormond Street Hospital Biomedical Research Centre. This work used services from the eHealth Core Facility at Karolinska Institutet, which is supported by the Strategic Research Area Healthcare Science (SFO-V). This research was funded by the NIHR Heath Technology Assessment (HTA) (Ref 16/19/02). The views expressed are those of the author(s) and not necessarily those of the NHS, the NIHR or the Department of Health. C.H., C.L.H., E.B.D. and C.G. acknowledge the financial support of the NIHR Nottingham Biomedical Research Centre and NIHR MindTech MedTech Co-operative. The study sponsor and funders have no role in study design, including collection, management, analysis and interpretation of data; writing of the report; and the decision to submit the report for publication. The corresponding author (C.H.) had full access to all the data in the study and had final responsibility for the decision to submit for publication. Aside from receiving funding from NIHR to support their salaries C.L.H., L.M., E.B.D., R.J., K.K., B.J.B., C.S., C.M., E.S., P.A., I.H., J.K., R.H., M.L.N., A.E., L.R.C., N.K., S.B., C.G. and E.M. declare no conflict of interest. C.H. declares he was the principal investigator on a grant from the National Institute of Health Research (NIHR) Health Technology Assessment programme to conduct an Evidence Synthesis on the treatments for tics and Tourette syndrome in children and young people’ HTA Project:10/142/01. D.M-C. reports personal fees from UpToDate Inc. outside the submitted work. T.M. reports personal fees from Royalties from Jessica Kingsley Publishers received for the following publications: The Tourettes Survival Kit: Tools for Young Adults with Tics (2019) by Tara Murphy and Damon Millar personal fees from Tic Disorders: A Guide for Parents and Professionals (2016) by Uttom Chowdhury and Tara Murphy from The Incredible Teenage Brain: Everything You Need to Know to Unlock Your Teen's Potential (2019) by Bettina Hohnen, Jane Gilmour & Tara Murphy, outside the submitted work. Funding Information: The authors also acknowledge that all research at . This work used services from the eHealth Core Facility at Karolinska Institutet, which is supported by the Strategic Research Area Healthcare Science (SFO‐V). Great Ormond Street Hospital NHS Foundation Trust and UCL Great Ormond Street Institute of Child Health is made possible by the NIHR Great Ormond Street Hospital Biomedical Research Centre Publisher Copyright: © 2023 The Authors. Journal of Child Psychology and Psychiatry published by John Wiley & Sons Ltd on behalf of Association for Child and Adolescent Mental Health.

PY - 2023/6

Y1 - 2023/6

N2 - Background: Little is known about the long-term effectiveness of behavioural therapy for tics. We aimed to assess the long-term clinical and cost-effectiveness of online therapist-supported exposure and response prevention (ERP) therapy for tics 12 and 18 months after treatment initiation. Methods: ORBIT (online remote behavioural intervention for tics) was a two-arm (1:1 ratio), superiority, single-blind, multicentre randomised controlled trial comparing online ERP for tics with online psychoeducation. The trial was conducted across two Child and Adolescent Mental Health Services in England. Participants were recruited from these two sites, across other clinics in England, or by self-referral. This study was a naturalistic follow-up of participants at 12- and 18-month postrandomisation. Participants were permitted to use alternative treatments recommended by their clinician. The key outcome was the Yale Global Tic Severity Scale Total Tic Severity Score (YGTSS-TTSS). A full economic evaluation was conducted. Registrations are ISRCTN (ISRCTN70758207); ClinicalTrials.gov (NCT03483493). Results: Two hundred and twenty-four participants were enrolled: 112 to ERP and 112 to psychoeducation. The sample was predominately male (177; 79%) and of white ethnicity (195; 87%). The ERP intervention reduced baseline YGTSS-TTSS by 2.64 points (95% CI: −4.48 to −0.79) with an effect size of −0.36 (95% CI: −0.61 to −0.11) after 12 months and by 2.01 points (95% CI: −3.86 to −0.15) with an effect size of −0.27 (95% CI -0.52 to −0.02) after 18 months, compared with psychoeducation. Very few participants (<10%) started new tic treatment during follow-up. The cost difference in ERP compared with psychoeducation was £304.94 (−139.41 to 749.29). At 18 months, the cost per QALY gained was £16,708 for ERP compared with psychoeducation. Conclusions: Remotely delivered online ERP is a clinical and cost-effective intervention with durable benefits extending for up to 18 months. This represents an efficient public mental health approach to increase access to behavioural therapy and improve outcomes for tics.

AB - Background: Little is known about the long-term effectiveness of behavioural therapy for tics. We aimed to assess the long-term clinical and cost-effectiveness of online therapist-supported exposure and response prevention (ERP) therapy for tics 12 and 18 months after treatment initiation. Methods: ORBIT (online remote behavioural intervention for tics) was a two-arm (1:1 ratio), superiority, single-blind, multicentre randomised controlled trial comparing online ERP for tics with online psychoeducation. The trial was conducted across two Child and Adolescent Mental Health Services in England. Participants were recruited from these two sites, across other clinics in England, or by self-referral. This study was a naturalistic follow-up of participants at 12- and 18-month postrandomisation. Participants were permitted to use alternative treatments recommended by their clinician. The key outcome was the Yale Global Tic Severity Scale Total Tic Severity Score (YGTSS-TTSS). A full economic evaluation was conducted. Registrations are ISRCTN (ISRCTN70758207); ClinicalTrials.gov (NCT03483493). Results: Two hundred and twenty-four participants were enrolled: 112 to ERP and 112 to psychoeducation. The sample was predominately male (177; 79%) and of white ethnicity (195; 87%). The ERP intervention reduced baseline YGTSS-TTSS by 2.64 points (95% CI: −4.48 to −0.79) with an effect size of −0.36 (95% CI: −0.61 to −0.11) after 12 months and by 2.01 points (95% CI: −3.86 to −0.15) with an effect size of −0.27 (95% CI -0.52 to −0.02) after 18 months, compared with psychoeducation. Very few participants (<10%) started new tic treatment during follow-up. The cost difference in ERP compared with psychoeducation was £304.94 (−139.41 to 749.29). At 18 months, the cost per QALY gained was £16,708 for ERP compared with psychoeducation. Conclusions: Remotely delivered online ERP is a clinical and cost-effective intervention with durable benefits extending for up to 18 months. This represents an efficient public mental health approach to increase access to behavioural therapy and improve outcomes for tics.

UR - http://www.scopus.com/inward/record.url?scp=85146327788&partnerID=8YFLogxK

U2 - 10.1111/jcpp.13756

DO - 10.1111/jcpp.13756

M3 - Article

SN - 0021-9630

VL - 64

SP - 941

EP - 951

JO - Journal of child psychology and psychiatry

JF - Journal of child psychology and psychiatry

IS - 6

ER -

Hollis C, Hall CL, Khan K, Jones R, Marston L, Le novere M et al. Long‐term clinical and cost‐effectiveness of a therapist‐supported online remote behavioural intervention for tics in children and adolescents: extended 12‐ and 18‐month follow‐up of a single‐blind randomised controlled trial. Journal of child psychology and psychiatry. 2023 Jun;64(6):941-951. Epub 2023 Jan 17. doi: 10.1111/jcpp.13756

Long‐term clinical and cost‐effectiveness of a therapist‐supported online remote behavioural intervention for tics in children and adolescents: extended 12‐ and 18‐month follow‐up of a single‐blind randomised controlled trial

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