Comparison of cognitive behaviour therapy versus activity management, both delivered remotely, to treat paediatric chronic fatigue syndrome/myalgic encephalomyelitis: the UK FITNET-NHS RCT.
ADOLESCENT
CAREGIVERS
CHILD
CHRONIC FATIGUE SYNDROME
COGNITIVE BEHAVIORAL THERAPY
E-COUNSELLING
E-THERAPY
EHEALTH
MYALGIC ENCEPHALOMYELITIS
ONLINE SYSTEMS
PARENTS
PEDIATRICS
PILOT PROJECTS
PRAGMATIC CLINICAL TRIAL
QUALITATIVE RESEARCH
RANDOMIZED CONTROLLED TRIAL
YOUNG PERSON
Journal
Health technology assessment (Winchester, England)
ISSN: 2046-4924
Titre abrégé: Health Technol Assess
Pays: England
ID NLM: 9706284
Informations de publication
Date de publication:
Oct 2024
Oct 2024
Historique:
medline:
1
11
2024
pubmed:
1
11
2024
entrez:
1
11
2024
Statut:
ppublish
Résumé
Parallel-group randomised controlled trial. Adolescents aged 11-17 years, diagnosed with myalgic encephalomyelitis/chronic fatigue syndrome and with no local specialist treatment centre, were referred to a specialist service in South West England. Fatigue In Teenagers on the interNET in the National Health Service is a web-based myalgic encephalomyelitis/chronic fatigue syndrome-focused cognitive-behavioural therapy programme for adolescents, supported by individualised written, asynchronous electronic consultations with a clinical psychologist/cognitive-behavioural therapy practitioner. The comparator was videocall-delivered activity management with a myalgic encephalomyelitis/chronic fatigue syndrome clinician. Both treatments were intended to last 6 months. Estimate the effectiveness of Fatigue In Teenagers on the interNET in the National Health Service compared to Activity Management for paediatric myalgic encephalomyelitis/chronic fatigue syndrome. Estimate the effectiveness of Fatigue In Teenagers on the interNET in the National Health Service compared to Activity Management for those with mild/moderate comorbid mood disorders. From a National Health Service perspective, estimate the cost-effectiveness of Fatigue In Teenagers on the interNET in the National Health Service compared to Activity Management over a 12-month horizon. 36-item Short Form Health Survey Physical Function subscale at 6 months post randomisation. Web-based, using minimisation with a random component to balance allocated groups by age and gender. While the investigators were blinded to group assignment, this was not possible for participants, parents/carers and therapists. The treatment of 314 adolescents was randomly allocated, 155 to Fatigue In Teenagers on the interNET in the National Health Service. Mean age was 14 years old and 63% were female. At 6 months, participants allocated to Fatigue In Teenagers on the interNET in the National Health Service were more likely to have improved physical function (mean 60.5, standard deviation 29.5, Fatigue In Teenagers on the interNET in the National Health Service participants attended, on average, half a day more school per week at 6 months than those allocated Activity Management, and this difference was maintained at 12 months. There was no strong evidence that comorbid mood disorder impacted upon the relative effectiveness of the two interventions. Similar improvement was seen in the two groups for pain and the Clinical Global Impression scale, with a mixed picture for fatigue. Both groups continued to improve, and no clear difference in physical function remained at 12 months [difference in means 4.4 (95% confidence interval -1.7 to 10.5)]. One or more of the pre-defined measures of a worsening condition in participants during treatment, combining therapist and patient reports, were met by 39 (25%) participants in the Fatigue In Teenagers on the interNET in the National Health Service group and 42 (26%) participants in the Activity Management group. A small gain was observed for the Fatigue In Teenagers on the interNET in the National Health Service group compared to Activity Management in quality-adjusted life-years (0.002, 95% confidence interval -0.041 to 0.045). From an National Health Service perspective, the costs were £1047.51 greater in the Fatigue In Teenagers on the interNET in the National Health Service group (95% confidence interval £624.61 to £1470.41). At a base cost-effectiveness threshold of £20,000 per quality-adjusted life-year, the incremental cost-effectiveness ratio was £457,721 with incremental net benefit of -£1001 (95% confidence interval -£2041 to £38). At 6 months post randomisation, compared with Activity Management, Fatigue In Teenagers on the interNET in the National Health Service improved physical function and school attendance. The additional cost of Fatigue In Teenagers on the interNET in the National Health Service and limited sustained impact mean it is unlikely to be cost-effective. This trial is registered as ISRCTN18020851. This award was funded by the National Institute for Health and Care Research (NIHR) Health Technology Assessment programme (NIHR award ref: 14/192/109) and is published in full in Why did we do the study? The best evidence for the treatment of adolescents with myalgic encephalomyelitis/chronic fatigue syndrome is cognitive–behavioural therapy for fatigue delivered in person. In the United Kingdom, most adolescents with myalgic encephalomyelitis/chronic fatigue syndrome cannot get this specialist treatment where they live. Fatigue In Teenagers on the interNET in the National Health Service is an online treatment using cognitive–behavioural therapy designed for myalgic encephalomyelitis/chronic fatigue syndrome, which has been shown to work in the Netherlands. To find out if Fatigue In Teenagers on the interNET in the National Health Service would be beneficial in the United Kingdom, we compared Fatigue In Teenagers on the interNET in the National Health Service to Activity Management. Activity Management is the treatment most often offered to children and young people with myalgic encephalomyelitis/chronic fatigue syndrome in the United Kingdom, and aims to avoid peaks in activity (sometimes called ‘pacing’). What was the question? Does Fatigue In Teenagers on the interNET in the National Health Service lead to greater improvements in children and young people with myalgic encephalomyelitis/chronic fatigue syndrome when compared to Activity Management, when both interventions are delivered remotely? What did we do? We compared Fatigue In Teenagers on the interNET in the National Health Service and Activity Management in two comparable groups of children, and measured physical function at 6 months as the main indication of improvement. We measured how much the treatments cost and we asked children and young people, their parents and treatment providers what they thought about the two interventions. What did we find? At 6 months, adolescents saw greater improvements in physical function, and attended half a day more school per week, with Fatigue In Teenagers on the interNET in the National Health Service compared to Activity Management. Both interventions were associated with improvements over 12 months, with there being no clear difference between them after that time. However, the Fatigue In Teenagers on the interNET in the National Health Service treatment was more expensive. What does this mean? We have shown that cognitive–behavioural therapy for fatigue can be provided online to children as Fatigue In Teenagers on the interNET in the National Health Service, leading to faster improvement in physical function and greater school attendance compared to Activity Management. However, Fatigue In Teenagers on the interNET in the National Health Service is expensive and is unlikely to be good value for money.
Autres résumés
Type: plain-language-summary
(eng)
Why did we do the study? The best evidence for the treatment of adolescents with myalgic encephalomyelitis/chronic fatigue syndrome is cognitive–behavioural therapy for fatigue delivered in person. In the United Kingdom, most adolescents with myalgic encephalomyelitis/chronic fatigue syndrome cannot get this specialist treatment where they live. Fatigue In Teenagers on the interNET in the National Health Service is an online treatment using cognitive–behavioural therapy designed for myalgic encephalomyelitis/chronic fatigue syndrome, which has been shown to work in the Netherlands. To find out if Fatigue In Teenagers on the interNET in the National Health Service would be beneficial in the United Kingdom, we compared Fatigue In Teenagers on the interNET in the National Health Service to Activity Management. Activity Management is the treatment most often offered to children and young people with myalgic encephalomyelitis/chronic fatigue syndrome in the United Kingdom, and aims to avoid peaks in activity (sometimes called ‘pacing’). What was the question? Does Fatigue In Teenagers on the interNET in the National Health Service lead to greater improvements in children and young people with myalgic encephalomyelitis/chronic fatigue syndrome when compared to Activity Management, when both interventions are delivered remotely? What did we do? We compared Fatigue In Teenagers on the interNET in the National Health Service and Activity Management in two comparable groups of children, and measured physical function at 6 months as the main indication of improvement. We measured how much the treatments cost and we asked children and young people, their parents and treatment providers what they thought about the two interventions. What did we find? At 6 months, adolescents saw greater improvements in physical function, and attended half a day more school per week, with Fatigue In Teenagers on the interNET in the National Health Service compared to Activity Management. Both interventions were associated with improvements over 12 months, with there being no clear difference between them after that time. However, the Fatigue In Teenagers on the interNET in the National Health Service treatment was more expensive. What does this mean? We have shown that cognitive–behavioural therapy for fatigue can be provided online to children as Fatigue In Teenagers on the interNET in the National Health Service, leading to faster improvement in physical function and greater school attendance compared to Activity Management. However, Fatigue In Teenagers on the interNET in the National Health Service is expensive and is unlikely to be good value for money.
Types de publication
Journal Article
Randomized Controlled Trial
Langues
eng
Sous-ensembles de citation
IM
Pagination
1-134Références
Lim E-J, Ahn Y-C, Jang E-S, Lee S-W, Lee S-H, Son C-G. Systematic review and meta-analysis of the prevalence of chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME). J Transl Med 2020;18(1):1–15.
Royal College of P, Child H. Evidence Based Guideline for the Management of CFS/ME (Chronic Fatigue Syndrome/Myalgic Encephalopathy) in Children and Young People. London: Royal College of Paediatrics and Child Health; 2004.
Baker R, Shaw B. Guidelines: Diagnosis and management of chronic fatigue syndrome or myalgic encephalomyelitis (or encephalopathy): Summary of NICE guidance. BMJ 2007;335(7617):446–8. https://doi.org/10.1136/bmj.39302.509005.AE
National Institute for Health and Care Excellence (NICE). Myalgic Encephalomyelitis (or Encephalopathy)/chronic Fatigue Syndrome: Diagnosis and Management; 2021. URL: www.nice.org.uk/guidance/ng206 (accessed 24 March 2023).
Rangel L, Garralda ME, Levin M, Roberts H. The course of severe chronic fatigue syndrome in childhood. J R Soc Med 2000;93(3):129–34.
Crawley E, Sterne JAC. Association between school absence and physical function in paediatric chronic fatigue syndrome/myalgic encephalopathy. Arch Dis Child 2009;94(10):752–6.
Webb CM, Collin SM, Deave T, Haig-Ferguson A, Spatz A, Crawley E. What stops children with a chronic illness accessing health care: a mixed methods study in children with Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME). BMC Health Serv Res 2011;11(1):308.
Nijhof SL, Bleijenberg G, Uiterwaal CS, Kimpen JL, van de Putte EM. Effectiveness of internet-based cognitive behavioural treatment for adolescents with chronic fatigue syndrome (FITNET): a randomised controlled trial. Lancet 2012;379(9824):1412–8.
Cairns R, Hotopf M. A systematic review describing the prognosis of chronic fatigue syndrome. Occup Med (Oxf) 2005;55(1):20–31.
Missen A, Hollingworth W, Eaton N, Crawley E. The financial and psychological impacts on mothers of children with chronic fatigue syndrome (CFS/ME). Child Care Health Dev 2012;38(4):505–12.
White PD, Sharpe MC, Chalder T, DeCesare JC, Walwyn R, the P. Protocol for the PACE trial: A randomised controlled trial of adaptive pacing, cognitive behaviour therapy, and graded exercise as supplements to standardised specialist medical care versus standardised specialist medical care alone for patients with the chronic fatigue syndrome/myalgic encephalomyelitis or encephalopathy. BMC Neurol 2007;7(1):6.
Whiting P, Bagnall A-M, Sowden AJ, Cornell JE, Mulrow CD, Ramírez G. Interventions for the treatment and management of chronic fatigue syndrome a systematic review. JAMA 2001;286(11):1360–8.
Larun L, Brurberg KG, Odgaard‐Jensen J, Price JR. Exercise therapy for chronic fatigue syndrome. Cochrane Database Syst Rev 2019;10(10):CD003200.
Chambers D, Bagnall A-M, Hempel S, Forbes C. Interventions for the treatment, management and rehabilitation of patients with chronic fatigue syndrome/myalgic encephalomyelitis: an updated systematic review. J R Soc Med 2006;99(10):506–20.
Castell BD, Kazantzis N, Moss-Morris RE. Cognitive behavioral therapy and graded exercise for chronic fatigue syndrome: a meta‐analysis. Clin Psychol: Sci Pract 2011;18(4):311–24.
White PD, Goldsmith KA, Johnson AL, Potts L, Walwyn R, DeCesare JC, et al.; PACE trial management group. Comparison of adaptive pacing therapy, cognitive behaviour therapy, graded exercise therapy, and specialist medical care for chronic fatigue syndrome (PACE): a randomised trial. Lancet (London, England) 2011;377(9768):823–36.
Smith SN, Crawley E. Is there effective behavioural treatment for children with chronic fatigue syndrome/myalgic encephalomyelitis? Arch Dis Child 2013;98(7):561–3.
Moore Y, Anderson N, Crawley E. G358 A systematic review to identify the definitions of recovery for paediatric patients with chronic fatigue syndrome (cfs) or myalgic encephalomyelitis (me) used in studies since 1994. Arch Dis Child 2015;100(Suppl 3):A146.3–A147.
Knight SJ, Scheinberg A, Harvey AR. Interventions in pediatric chronic fatigue syndrome/myalgic encephalomyelitis: a systematic review. J Adolesc Health 2013;53(2):154–65.
Stulemeijer M, de Jong LW, Fiselier TJ, Hoogveld SW, Bleijenberg G. Cognitive behaviour therapy for adolescents with chronic fatigue syndrome: randomised controlled trial. BMJ 2004;330(7481):14.
Al-Haggar MS, Al-Naggar ZA, Abdel-Salam MA. Biofeedback and cognitive behavioral therapy for Egyptian adolescents suffering from chronic fatigue syndrome. J Pediatr Neurol 2006;4(03):161–9.
Chalder T, Deary V, Husain K, Walwyn R. Family-focused cognitive behaviour therapy versus psycho-education for chronic fatigue syndrome in 11-to 18-year-olds: a randomized controlled treatment trial. Psychol Med 2010;40(8):1269–79.
Albers E, Nijhof LN, Berkelbach van der Sprenkel EE, van de Putte EM, Nijhof SL, Knoop H. Effectiveness of internet-based cognitive behavior therapy (Fatigue In Teenagers on the interNET) for adolescents with chronic fatigue syndrome in routine clinical care: observational study. J Med Internet Res 2021;23(8):e24839.
Bould H, Collin SM, Lewis G, Rimes K, Crawley E. Depression in paediatric chronic fatigue syndrome. Arch Dis Child 2013;98(6):425–8.
Crawley E, Hunt L, Stallard P. Anxiety in children with CFS/ME. Eur Child Adolesc Psychiatry 2009;18(11):683–9.
Loades ME, Rimes KA, Ali S, Chalder T. Depressive symptoms in adolescents with chronic fatigue syndrome (CFS): Are rates higher than in controls and do depressive symptoms affect outcome? Clin Child Psychol Psychiatry 2019;24(3):580–92.
Prins J, Bleijenberg G, Rouweler EK, Van Der Meer J. Effect of psychiatric disorders on outcome of cognitive-behavioural therapy for chronic fatigue syndrome. Br J Psychiatry 2005;187(2):184–5.
Kempke S, Goossens L, Luyten P, Bekaert P, Van Houdenhove B, Van Wambeke P. Predictors of outcome in a multi-component treatment program for chronic fatigue syndrome. J Affect Disord 2010;126(1-2):174–9.
Tummers M, Knoop H, van Dam A, Bleijenberg G. Moderators of the treatment response to guided self-instruction for chronic fatigue syndrome. J Psychosom Res 2013;74(5):373–7.
Loades ME, Stallard P, Morris R, Kessler D, Crawley E. Do adolescents with chronic fatigue syndrome (CFS/ME) and co-morbid anxiety and/or depressive symptoms think differently to those who do not have co-morbid psychopathology? J Affect Disord 2020;274:752–8.
Health NCCfM. E-therapies Systematic Review for Children and Young People with Mental Health Problems. The British Psychological Society and The Royal College of Psychiatrists; 2014.
Cervin M, Lundgren T. Technology-delivered cognitive-behavioral therapy for pediatric anxiety disorders: a meta-analysis of remission, posttreatment anxiety, and functioning. J Child Psychol Psychiatry 2022;63(1):7–18.
Fisher E, Law E, Dudeney J, Eccleston C, Palermo TM. Psychological therapies (remotely delivered) for the management of chronic and recurrent pain in children and adolescents. Cochrane Database Syst Rev 2019;(4):CD011118.
Baos S, Brigden A, Anderson E, Hollingworth W, Price S, Mills N, et al. Investigating the effectiveness and cost-effectiveness of FITNET-NHS (Fatigue In Teenagers on the interNET in the NHS) compared to Activity Management to treat paediatric chronic fatigue syndrome (CFS)/myalgic encephalomyelitis (ME): protocol for a randomised controlled trial. Trials 2018;19(1):1–12.
Anderson E, Gaunt D, Metcalfe C, Rai M, Hollingworth W, Mills N, et al. Investigating the effectiveness and cost-effectiveness of FITNET-NHS (Fatigue In Teenagers on the interNET in the NHS) compared to activity management to treat paediatric chronic fatigue syndrome (CFS)/myalgic encephalomyelitis (ME): amendment to the published protocol. Trials 2019;20(1):1–3.
Esbjørn BH, Sømhovd MJ, Turnstedt C, Reinholdt-Dunne ML. Assessing the Revised Child Anxiety and Depression Scale (RCADS) in a national sample of Danish youth aged 8–16 years. PLOS ONE 2012;7(5):e37339.
Harris PA, Taylor R, Thielke R, Payne J, Gonzalez N, Conde JG. Research electronic data capture (REDCap)—a metadata-driven methodology and workflow process for providing translational research informatics support. J Biomed Inform 2009;42(2):377–81.
Chorpita BF, Moffitt CE, Gray J. Psychometric properties of the Revised Child Anxiety and Depression Scale in a clinical sample. Behav Res Ther 2005;43(3):309–22.
Chorpita BF, Yim L, Moffitt C, Umemoto LA, Francis SE. Assessment of symptoms of DSM-IV anxiety and depression in children: a revised child anxiety and depression scale. Behav Res Ther 2000;38(8):835–55.
Bruce F, Chorpita CE, Susan H. Spence. Revised Children’s Anxiety and Depression Scale: User’s Guide; 2015. URL: www.childfirst.ucla.edu/wp-content/uploads/sites/163/2018/03/RCADSUsersGuide20150701.pdf (accessed 24 March 2023).
Hoffmann TC, Glasziou PP, Boutron I, Milne R, Perera R, Moher D, et al. Better reporting of interventions: template for intervention description and replication (TIDieR) checklist and guide. BMJ 2014;348:g1687.
Ware Jr JE, Sherbourne CD. The MOS 36-item short-form health survey (SF-36): I Conceptual framework and item selection. Med Care 1992;30:473–83.
Chalder T, Berelowitz G, Pawlikowska T, Watts L, Wessely S, Wright D, Wallace EP. Development of a fatigue scale. J Psychosom Res 1993;37(2):147–53.
Beurskens AJ, Bültmann U, Kant I, Vercoulen JH, Bleijenberg G, Swaen GM. Fatigue among working people: validity of a questionnaire measure. Occup Environ Med 2000;57(5):353–7.
Hawker GA, Mian S, Kendzerska T, French M. Measures of adult pain: Visual analog scale for pain (VAS pain), numeric rating scale for pain (NRS pain), McGill pain questionnaire (MPQ), short‐form McGill pain questionnaire (SF-MPQ), chronic pain grade scale (CPGS), short form‐36 bodily pain scale (SF‐36 BPS), and measure of intermittent and constant osteoarthritis pain (ICOAP). Arthritis Care Res 2011;63(S11):S240–52.
Busner J, Targum SD. The clinical global impressions scale: applying a research tool in clinical practice. Psychiatry (Edgmont) 2007;4(7):28–37.
Wille N, Badia X, Bonsel G, Burström K, Cavrini G, Devlin N, et al. Development of the EQ-5D-Y: a child-friendly version of the EQ-5D. Qual Life Res 2010;19(6):875–6.
Reilly MC, Zbrozek AS, Dukes EM. The validity and reproducibility of a work productivity and activity impairment instrument. PharmacoEcon 1993;4(5):353–65.
Parslow R, Patel A, Beasant L, Haywood K, Johnson D, Crawley E. What matters to children with CFS/ME? A conceptual model as the first stage in developing a PROM. Arch Dis Child 2015;100(12):1141–7.
Crawley EM, Gaunt DM, Garfield K, Hollingworth W, Sterne JAC, Beasant L, et al. Clinical and cost-effectiveness of the Lightning Process in addition to specialist medical care for paediatric chronic fatigue syndrome: randomised controlled trial. Arch Dis Child 2018;103(2):155–64.
Crawley E, Mills N, Beasant L, Johnson D, Collin SM, Deans Z, et al. The feasibility and acceptability of conducting a trial of specialist medical care and the Lightning Process in children with chronic fatigue syndrome: feasibility randomized controlled trial (SMILE study). Trials 2013;14(1):415.
Chalder T, Goldsmith KA, White PD, Sharpe M, Pickles AR. Rehabilitative therapies for chronic fatigue syndrome: a secondary mediation analysis of the PACE trial. Lancet Psychiatry 2015;2(2):141–52.
Loades ME, Rimes K, Lievesley K, Ali S, Chalder T. Cognitive and behavioural responses to symptoms in adolescents with chronic fatigue syndrome: a case-control study nested within a cohort. Clin Child Psychol Psychiatry 2019; 24(3):564–79.
Brigden A, Parslow RM, Gaunt D, Collin SM, Jones A, Crawley E. Defining the minimally clinically important difference of the SF-36 physical function subscale for paediatric CFS/ME: triangulation using three different methods. Health Qual Life Out 2018;16(1):202.
Metcalfe C, Crawley E. FITNET NHS Statistical Analysis Plan. University of Bristol; 2021.
Groenwold RH, Donders ART, Roes KC, Harrell Jr FE, Moons KG. Dealing with missing outcome data in randomized trials and observational studies. Am J Epidemiol 2012;175(3):210–7.
White IR, Carpenter J, Horton NJ. A mean score method for sensitivity analysis to departures from the missing at random assumption in randomised trials. Statistica Sinica 2018;28(4):1985–2003.
Curtis L, Burns A. Unit Costs of Health and Social Care 2020. Canterbury: Personal Social Services Research Unit; 2020.
National Health Service. 2019/20 National Cost Collection Data Publication. 2020. URL: www.england.nhs.uk/publication/2019-20-national-cost-collection-data-publication/ (accessed 24 March 2023).
Office for National Statistics. Annual Survey of Hours and Earnings (ASHE); 2020. URL: www.ons.gov.uk/surveys/informationforbusinesses/businesssurveys/annualsurveyofhoursandearningsashe
Curtis LA. Unit Costs of Health and Social Care 2013. Personal Social Services Research Unit, University of Kent; 2013.
Pope C, Turnbull J, Jones J, Prichard J, Rowsell A, Halford S. Has the NHS 111 urgent care telephone service been a success? Case study and secondary data analysis in England. BMJ Open 2017;7(5):e014815.
National Health Service Business Services Authority. Prescription Cost Analysis – England 2020/21; 2021. URL: www.nhsbsa.nhs.uk/statistical-collections/prescription-cost-analysis-england/prescription-cost-analysis-england-202021 (accessed 24 March 2023).
HM Revenue & Customs. Travel — Mileage and Fuel Rates and Allowances; 2022. URL: www.gov.uk/government/publications/rates-and-allowances-travel-mileage-and-fuel-allowances/travel-mileage-and-fuel-rates-and-allowances (accessed 24 March 2023).
Kind P, Klose K, Gusi N, Olivares PR, Greiner W. Can adult weights be used to value child health states? Testing the influence of perspective in valuing EQ-5D-Y. Qual Life Res 2015;24(10):2519–39.
Kreimeier S, Mott D, Ludwig K, Greiner W, Prevolnik Rupel V, Ramos-Goñi JM. EQ-5D-Y Value Set for Germany. PharmacoEcon 2022;40:217–29.
Manca A, Hawkins N, Sculpher MJ. Estimating mean QALYs in trial‐based cost‐effectiveness analysis: the importance of controlling for baseline utility. Health Econ 2005;14(5):487–96.
Hoch JS, Hay A, Isaranuwatchai W, Thavorn K, Leighl NB, Tu D, et al. Advantages of the net benefit regression framework for trial-based economic evaluations of cancer treatments: an example from the Canadian Cancer Trials Group CO 17 trial. BMC Cancer 2019;19(1):1–9.
Leurent B, Gomes M, Faria R, Morris S, Grieve R, Carpenter JR. Sensitivity analysis for not-at-random missing data in trial-based cost-effectiveness analysis: a tutorial. PharmacoEcon 2018;36(8):889–901.
Ramos-Goñi JM, Oppe M, Estévez-Carrillo A, Rivero-Arias O; IMPACT HTA HRQoL Group. Accounting for unobservable preference heterogeneity and evaluating alternative anchoring approaches to estimate country-specific EQ-5D-Y value sets: a case study using Spanish preference data. Value Health 2022;25(5):835–43.
Donovan JL, Rooshenas L, Jepson M, Elliott D, Wade J, Avery K, et al. Optimising recruitment and informed consent in randomised controlled trials: the development and implementation of the Quintet Recruitment Intervention (QRI). Trials 2016;17(1):283.
Donovan JL, Paramasivan S, de Salis I, Toerien M. Clear obstacles and hidden challenges: understanding recruiter perspectives in six pragmatic randomised controlled trials. Trials 2014;15(1):5.
Ritchie J, Lewis J, Nicholls CM, Ormston R. Qualitative Research Practice: A Guide for Social Science Students and Researchers. Sage; 2013.
Pope C, Ziebland S, Mays N. Analysing qualitative data. BMJ 2000;320(7227):114–6.
Glaser BG, Holton J. Discovery of Grounded Theory; 1967.
Braun V, Clarke V. Using thematic analysis in psychology. Qual Res Psychol 2006;3(2):77–101.
Guest G, Bunce A, Johnson L. How many interviews are enough?: An experiment with data saturation and variability. Field Methods 2006;18(1):59–82.
Anderson E, Parslow R, Hollingworth W, Mills N, Beasant L, Gaunt D, et al. Recruiting adolescents with chronic fatigue syndrome/myalgic encephalomyelitis to internet-delivered therapy: internal pilot within a randomized controlled trial. J Med Internet Res 2020;22(8):e17768.
Centre for Academic Child Health. FITNET-NHS Study – Overview; 2016. URL: www.bristol.ac.uk/academic-child-health/research/research/cfsme/fitnet-nhs/fitnet-nhs/ (accessed 23 March 2023).
Mills N, Blazeby JM, Hamdy FC, Neal DE, Campbell B, Wilson C, et al. Training recruiters to randomized trials to facilitate recruitment and informed consent by exploring patients’ treatment preferences. Trials 2014;15(1):1–13.
Mills N, Donovan JL, Wade J, Hamdy FC, Neal DE, Lane JA. Exploring treatment preferences facilitated recruitment to randomized controlled trials. J Clin Epidemiol 2011;64(10):1127–36.
Jones LS, Anderson E, Loades M, Barnes R, Crawley E. Can linguistic analysis be used to identify whether adolescents with a chronic illness are depressed? Clin Psychol Psychother 2020;27(2):179–92.
Office for National Statistics. Ethnic group by age and sex, England and Wales: Census 2021; 2023. URL: www.ons.gov.uk/peoplepopulationandcommunity/culturalidentity/ethnicity/articles/ethnicgroupbyageandsexenglandandwales/latest (accessed 24 March).
Lievesley K, Rimes KA, Chalder T. A review of the predisposing, precipitating and perpetuating factors in Chronic Fatigue Syndrome in children and adolescents. Clin Psychol Rev 2014;34(3):233–48.
Aisbitt GM, Nolte T, Fonagy P. Editorial Perspective: the digital divide - inequalities in remote therapy for children and adolescents. Child Adolesc Ment Health 2023;28(1):105–7.
Meyerowitz-Katz G, Ravi S, Arnolda L, Feng X, Maberly G, Astell-Burt T. Rates of attrition and dropout in app-based interventions for chronic disease: systematic review and meta-analysis. J Med Internet Res 2020;22(9):e20283.
Heins MJ, Knoop H, Prins JB, Stulemeijer M, van der Meer JWM, Bleijenberg G. Possible detrimental effects of cognitive behaviour therapy for chronic fatigue syndrome. Psychother Psychosom 2010;79(4):249–56.
McCrone P, Sharpe M, Chalder T, Knapp M, Johnson AL, Goldsmith KA, White PD. Adaptive pacing, cognitive behaviour therapy, graded exercise, and specialist medical care for chronic fatigue syndrome: a cost-effectiveness analysis. PLOS ONE 2012;7(8):e40808.
Vos-Vromans D, Evers S, Huijnen I, Köke A, Hitters M, Rijnders N, et al. Economic evaluation of multidisciplinary rehabilitation treatment versus cognitive behavioural therapy for patients with chronic fatigue syndrome: a randomized controlled trial. PLOS ONE 2017;12(6):e0177260.
Severens JL, Prins JB, van der Wilt GJ, van der Meer JW, Bleijenberg G. Cost-effectiveness of cognitive behaviour therapy for patients with chronic fatigue syndrome. QJM 2004;97(3):153–61.
Myers C, Wilks D. Comparison of Euroqol EQ-5D and SF-36 in patients with chronic fatigue syndrome. Qual Life Res 1999;8(1):9–16.
Spronk I, Polinder S, Bonsel G, Janssen M, Haagsma J. The relation between EQ-5D and fatigue in a Dutch general population sample: an explorative study. Health Qual Life Out 2021;19(1):1–11.
Cochrane M, Mitchell E, Hollingworth W, Crawley E, Trépel D. Cost-effectiveness of interventions for chronic fatigue syndrome or Myalgic encephalomyelitis: a systematic review of economic evaluations. Appl Health Econ Health Policy 2021;19(4):473–86.
Parslow RM, Shaw A, Haywood KL, Crawley E. Developing and pretesting a new patient reported outcome measure for paediatric Chronic Fatigue Syndrome/ Myalgic Encephalopathy (CFS/ME): cognitive interviews with children. J Patient Rep Outcomes 2019;3(1):67.
Gargon E, Gurung B, Medley N, Altman DG, Blazeby JM, Clarke M, Williamson PR. Choosing important health outcomes for comparative effectiveness research: a systematic review. PLOS ONE 2014;9(6):e99111.
Brueton VC, Stevenson F, Vale CL, Stenning SP, Tierney JF, Harding S, et al. Use of strategies to improve retention in primary care randomised trials: a qualitative study with in-depth interviews. BMJ Open 2014;4(1):e003835.
Kearney A, Rosala-Hallas A, Bacon N, Daykin A, Shaw ARG, Lane AJ, et al. Reducing attrition within clinical trials: the communication of retention and withdrawal within patient information leaflets. PLOS ONE 2018;13(10):e0204886.
Schneider LH, Hadjistavropoulos HD, Faller YN. Internet-delivered cognitive behaviour therapy for depressive symptoms: an exploratory examination of therapist behaviours and their relationship to outcome and therapeutic alliance. Behav Cogn Psychother 2016;44(6):625–39.
Paxling B, Lundgren S, Norman A, Almlov J, Carlbring P, Cuijpers P, Andersson G. Therapist behaviours in internet-delivered cognitive behaviour therapy: analyses of e-mail correspondence in the treatment of generalized anxiety disorder. Behav Cogn Psychother 2013;41:280–89. https://doi.org/10.1017/s1352465812000240