We thank all participants who have taken part throughout the programme; Kathy Rowan as independent chairperson of both the NIHR Programme Grant Committee and Trial Steering Committee for the ACtiF Programme; the members of the Independent Trial Steering Committee (including David Torgerson); the members of the PPI panel (including Caroline Greenslade, Martin Cameron, Karen Leyland and Ruth Day); trial support officers (Louise Turner and Helen Wakefield); the data management support team (Amanda Loban and Chris Turtle); the CFHealthHub support team (including Steven Antrobus); the PARI GmbH support team; the Sheffield Teaching Hospital as sponsor; Carla Girling for support in the PMGs; Hannah Cantrill for contributions as a research assistant; Susan Kirkpatrick for her contribution to WP 2.1C; Alexander Scott and Samuel Keating for their contributions to the qualitative work in WP 3.1; Saleema Rex for her contribution to the mediation analysis in WP 3.3; Kate Ennis for her contribution to the health economic evaluation in WP 3.5; pilot trial principal investigators (Julia Nightingale, Jane Dewar and Mark Allenby) and interventionists (including Claire Oliver); and RCT principal investigators (Edward Nash, Jo Whitehouse, Ian Ketchall, Jackie Rendall, Helen Rodgers, Caroline Elston, Stephen Bourke, William Flight, Angela McGowen, Danie Watson, Rebecca Thomas, Dejene Shiferaw, Kathryn Bateman, Nick Bell, Nick Withers, Chris Sheldon, David Derry, David Waine, Uta Hill, Jonathan Myers, Chandra Ohri, Giles Fitch and Susan Madge) and interventionists (Katherine Miles, Lisa Kent, Valerie Mills, Peter Moran, Georgia King, Lucy Funnell, Jocelyn Choyce, Jessica Williams, Cally Evans, Kamilla Dack, Jayne Trott, Annant Damani, Shehnaz Raniwalla, Darren Tature, Alan Anderson, Penny Galey, Louise Warnock, Jayne Faulkner, Kathryn Barbour, Michelle Thomas, Harriet Gledhill, Katherine Donohue, Heather Seabridge, Michael Martin,, Katy Lee, Corina Weir, Laura Barlow, Sarah Salberg and Caroline Whitton).
Contributions of authors
Martin J Wildman (https://orcid.org/0000-0002-1658-881X) (Consultant in Respiratory Medicine and Adult Cystic Fibrosis) was the joint chief investigator of the ACtiF programme.
Alicia O’Cathain (https://orcid.org/0000-0003-4033-506X) (Professor of Health Services Research) was the joint chief investigator of the ACtiF programme and led the qualitative research and process evaluations
Daniel Hind (https://orcid.org/0000-0002-6409-4793) (Professor in Complex Interventions) was a co-applicant, led the feasibility study and provided oversight for the management of the programme.
Chin Maguire (https://orcid.org/0000-0002-9397-7608) (Study Manager) was the programme manager.
Madelynne A Arden (https://orcid.org/0000-0002-6199-717X) (Professor of Health Psychology) was a co-applicant and led the design of the CFHealthHub intervention and development and delivery of the intervention training programme and input into the design and analysis of the fidelity assessment.
Marlene Hutchings (https://orcid.org/0000-0002-4710-657X) (Lead Physiotherapist) led WP 2.1C and worked closely with Madelynne Arden in the design of the CFHealthHub intervention and development and delivery of the intervention training programme and fidelity assessment.
Judy Bradley (https://orcid.org/0000-0002-7423-135X) (Professor of Respiratory Health) led the design and analysis of the fidelity assessment and worked with Madelynne A Arden and Marlene Hutchings to deliver the intervention training programme.
Stephen J Walters (https://orcid.org/0000-0001-9000-8126) (Professor of Medical Statistics and Clinical Trials) led the statistical analysis throughout the programme.
Pauline Whelan (https://orcid.org/0000-0001-8689-3919) (Information Systems Manager) led the technical development of the CFHealthHub digital platform throughout the programme.
John Ainsworth (https://orcid.org/0000-0002-2187-9195) (Professor of Health Informatics) was a co-applicant and provided oversight of the technical development of the CFHealthHub digital platform.
Paul Tappenden (https://orcid.org/0000-0001-6612-2332) (Professor of Health Economic Modelling) was a co-applicant and led the health economic evaluation in WP 3.5.
Iain Buchan (https://orcid.org/0000-0003-3392-1650) (Chair in Public Health and Clinical Informatics for Population Health Sciences) was a co-applicant and advised on the digital aspects of data acquisition.
Rachel Elliott (https://orcid.org/0000-0002-3650-0168) (Professor in Health Economics) was a co-applicant and provided input to the health economic evaluation.
John Nicholl (https://orcid.org/0000-0001-5436-1264) (Professor of Health Services Research) was a co-applicant and advised on the RCT design.
Stuart Elborn (https://orcid.org/0000-0002-2323-442X) (Professor of Respiratory Medicine) was a co-applicant and advised on interpretation of the results from a clinical CF perspective.
Susan Michie (https://orcid.org/0000-0003-0063-6378) (Professor of Health Psychology) was a co-applicant and advised on developing the BCI.
Laura Mandefield (https://orcid.org/0000-0002-4219-5673) (Research Fellow/Statistician) conducted statistical analyses.
Laura Sutton (https://orcid.org/0000-0003-3327-5927) (Research Associate/Statistician) conducted statistical analyses.
Zhe Hui Hoo (https://orcid.org/0000-0002-7067-3783) (Clinical Research Fellow) provided ongoing clinical expertise.
Sarah J Drabble (https://orcid.org/0000-0001-7183-6321) (Research Fellow) led and conducted the qualitative data collection and analysis.
Elizabeth Lumley (https://orcid.org/0000-0002-8962-7568) (Research Associate) conducted qualitative data collection and analysis in WP 3.
Daniel Beever (https://orcid.org/0000-0001-9063-3677) (Study Manager and PPI lead) was a co-applicant and stood as PPI chairperson throughout the programme.
Aline Navega Biz (https://orcid.org/0000-0002-6923-4556) (Research Associate) conducted the health economic evaluation with Paul Tappenden in WP 3.5.
Anne Scott (https://orcid.org/0000-0003-4396-438X) (Research Assistant) led the context analysis conducted in WP 3.3, contributed to qualitative analyses and worked in the programme management team.
Simon Waterhouse (https://orcid.org/0000-0002-6303-9610) (Data Specialist) led data management activity and critical analyses that enabled the calculation of adherence outcomes.
Louisa Robinson (https://orcid.org/0000-0001-5680-0734) (Research Assistant) led the drafting of the report, worked in the programme management team and provided analyses for WP 2.3 and 3.3.
Mónica Hernández Alava (https://orcid.org/0000-0003-4474-5883) (Reader in Health Econometrics) conducted the health economic evaluation with Paul Tappenden in WP 3.5.
Alessandro Sasso (https://orcid.org/0000-0001-8909-926X) (Research Associate, Health Econometrician) conducted the health economic evaluation with Paul Tappenden in WP 3.5.
All authors contributed to the drafting of this report and had the opportunity to approve the final version.
Publications
Peer-reviewed journal articles published
Hoo ZH, Curley R, Campbell MJ, Walters SJ, Hind D, Wildman MJ. Accurate reporting of adherence to inhaled therapies in adults with cystic fibrosis: methods to calculate ‘normative adherence’. Patient Prefer Adherence 2016;10:887–900.
Tappenden P, Sadler S, Wildman M. An early health economic analysis of the potential cost effectiveness of an adherence intervention to improve outcomes for patients with cystic fibrosis. PharmacoEconomics 2017;35:647–59.
Hoo ZH, Coates E, Maguire C, Cantrill H, Shafi N, Nash EF, et al. Pragmatic criteria to define chronic pseudomonas aeruginosa infection among adults with cystic fibrosis. Eur J Clin Microbiol Infect Dis 2018;37:2219–22.
Arden MA, Drabble S, O’Cathain A, Hutchings M, Wildman M. Adherence to medication in adults with cystic fibrosis: an investigation using objective adherence data and the theoretical domains framework. Br J Health Psychol 2019;24:357–80.
Drabble SJ, O’Cathain A, Arden MA, Hutchings M, Beever D, Wildman M. When is forgetting not forgetting? A discursive analysis of differences in forgetting talk between adults with cystic fibrosis with different levels of adherence to nebulizer treatments. QualHealthRes 2019;29:2119–31.
Gao J, Arden M, Hoo ZH, Wildman M. Understanding patient activation and adherence to nebuliser treatment in adults with cystic fibrosis: responses to the UK version of PAM-13 and a think aloud study. BMC Health Serv Res 2019;19:420.
Hind D, Drabble SJ, Arden MA, Mandefield L, Waterhouse S, Maguire C, et al. Supporting medication adherence for adults with cystic fibrosis: a randomised feasibility study. BMC Pulm Med 2019;19:77.
Hoo ZH, Gardner B, Arden MA, Waterhouse S, Walters SJ, Campbell MJ, et al. Role of habit in treatment adherence among adults with cystic fibrosis. Thorax 2019;74:197–9.
Hoo ZH, Wildman MJ, Campbell MJ, Walters SJ, Gardner B. A pragmatic behavior-based habit index for adherence to nebulized treatments among adults with cystic fibrosis. Patient Prefer Adherence 2019;13:283–94.
Drabble SJ, O’Cathain A, Scott AJ, Arden MA, Keating S, Hutchings M, et al. Mechanisms of action of a web-based intervention with health professional support to increase adherence to nebulizer treatments in adults with cystic fibrosis: qualitative interview study. J Med Internet Res 2020;22:e16782.
Hind D, Drabble SJ, Arden MA, Mandefield L, Waterhouse S, Maguire C, et al. Feasibility study for supporting medication adherence for adults with cystic fibrosis: mixed-methods process evaluation. BMJ Open 2020;10:e039089.
Arden M, Hutchings M, Whelan P, Drabble SJ, Beever D, Bradley JM, et al. Development of an intervention to increase adherence to nebuliser treatment in adults with cystic fibrosis: CFHealthHub. Pilot and Feasibility Stud 2021;7:PMC7780635.
Wildman MJ, O’Cathain A, Maguire C, Arden MA, Hutchings M, Bradley J, et al. Self-management intervention to reduce pulmonary exacerbations by supporting treatment adherence in adults with cystic fibrosis: a randomised controlled trial [published online ahead of print September 23 2021]. Thorax 2021.
Conference abstracts
Arden MA, Drabble SJ, O’Cathain A, Hutchings M, Wildman M. ACtiF study: understanding adherence to nebuliser treatment in adults with cystic fibrosis using the theoretical domains framework. J Cyst Fibros 2016;15:S26.
Arden MA, Drabble SJ, O’Cathain A, Hutchings M, Wildman M. Applying the theoretical domains framework to adherence to nebuliser treatment in adults with cystic fibrosis. Eur Heal Psychol 2016;18:689.
Sadler S, Wildman M, Tappenden P. Cost-effectiveness of a complex adherence intervention to improve prevention in cystic fibrosis by empowering effective self-management: an exploratory health economic evaluation using UK Registry data. J Cyst Fibros 2016;15:S119–20.
Whelan P. Co-producing Digital Platforms to Support an Ageing Population, Employing Information and Communications Technologies in Homes and Cities for the Health and Well-Being of Older People, Sichuan University, Chengdu, China, 15–17 August 2016.
Whelan P. Using Digital Platforms to Improve the Lives of People with Chronic Conditions: A Connected Health Case Study of CFHealthHub, Mobile Apps & Behaviour Change for Connected Health Seminar, University of Manchester, Manchester, UK, 27 September 2016.
Wildman M, Buchan I, Ainsworth J, Whelan, P. CFHealthHub: Complex Intervention to Support Self-Management in People with Cystic Fibrosis. A Case Study for the Farr Institute ‘100 Ways of Using Data to Make Lives Better’. West De Moines, IA: Farr Institute. 2016.
Arden M, Drabble S, O’Cathain A, Hind D, Hutchings M, Whelan P, et al. CFHealthHub: The Development of a Digital Intervention to Provide Feedback on Objective Nebuliser Adherence Data for Adults with Cystic Fibrosis (CF). 3rd UCL Centre for Behaviour Change Digital Health Conference 2017: Harnessing Digital Technology for Behaviour Change, 22–23 February 2017, London, UK.
Arden MA, Hutchings M, Whelan P, Wildman MJ, CFHealthHub group. Understanding Nebuliser Adherence in Adults with Cystic Fibrosis: Comparing High, Medium and Low Adherers. 31st European Health Psychologist conference, 31 August to 2 September 2017, Padua, Italy.
Hind D, Maguire C, Cantrill H, O’Cathain A, Wildman M. CFHealthHub: complex intervention to support adherence to treatment in adults with cystic fibrosis: external pilot trial. J Cyst Fibros 2017;16:S40–1.
Hoo ZH, Curley RE, Campbell SMJ, Wildman MJ. Cluster analysis of objective nebuliser adherence data among adults with CF: change in adherence over time and association with clinical outcomes. J Cyst Fibros 2017;16:S60–1.
Hoo ZH, Waterhouse S, Nightingale JA, Dewar J, Allenby M, Haynes F, et al. CFHealthHub: using Leeds criteria and clinicians’ decision to determine the pseudomonas status among the 64 adults with cystic fibrosis in the two centre CFHealthHub (CFHealthHub) pilot study. J Cyst Fibros 2017;16:S105–6.
Horspool K, Whelan P, Wildman M. CFHealthHub: iterative co-production of a data feedback platform providing patient level adherence data to multi-disciplinary teams to optimise individual care and centre level adherence data to centre directors allowing benchmarking to enable centre level quality improvement cycles in a 3 centre improvement collaborative. J Cyst Fibros 2017;16:S155.
Kirkpatrick S, Arden M, Beever D, Bradley J, Cantrill H, Daniels T, et al. CFHealthHub: development and evaluation of videos incorporating peer description of successful self-management with inhaled therapies in adults with CF used to build self-efficacy to support self-care within the CFHealthHub complex intervention. J Cyst Fibros 2017;16:S155–6.
Whelan P. Data and mHealth Technologies. Digital Health Outlook, UK and Italy Experiences in a Global Context, 20 March 2017, Milan, Italy.
Whelan P, Hassan L, Fox S, Tempest E. Developing Mobile Applications for Health. Informatics for Health Congress, 24–26 April 2017, Manchester, UK.
Arden MA, Gao J, Hoo ZH, Wildman M. The Patient Activation Measure and Adherence in Adults with Cystic Fibrosis: A Think-Aloud Study. BPS Division of Health Psychology Annual Conference, 5–7 September 2018, Newcastle upon Tyne, UK.
Wildman M, Hutchings M, Arden M, O’Cathain A, Drabble S, Walters SJ, et al. Development and Evaluation of an Intervention to Support Adherence to Treatment in Adults with Cystic Fibrosis: A Randomised Controlled Trial and Parallel Process Evaluation Protocol. Sheffield: Sheffield Teaching Hospitals NHS Foundation Trust; 2018.
Arden M. Health and habits. Psychologist 2019;32:7.
Whelan P, Stockton-Powdrell C, van Velsen L, Tabak M, Wolf K. Interdisciplinary Perspectives on Connected Health Research: Facilitators and Barriers for Sustainable Platform Development. MedInfo: 17th World Congress of Medical and Health Informatics, 25–30 August 2019, Lyon, France.
Other publications
Beever DA, Wildman M. A personal reflection on being a co-applicant in the ACtiF study. Res Involv Engagem 2017;3:12.
Data-sharing statement
Requests for patient-level data and statistical code should be made to the corresponding author and will be considered by members of the original trial management group, including the chief investigators and members of the Clinical Trials Research Unit (CTRU), who will release data on a case-by-case basis. Data will be shared following the principles for sharing patient-level data as described by Smith et al.86 The data will not contain any direct identifiers, we will minimise indirect identifiers and we will remove free-text data to minimise the risk of identification.
Patient data
This work uses data provided by patients and collected by the NHS as part of their care and support. Using patient data is vital to improve health and care for everyone. There is huge potential to make better use of information from people’s patient records, to understand more about disease, develop new treatments, monitor safety, and plan NHS services. Patient data should be kept safe and secure, to protect everyone’s privacy, and it’s important that there are safeguards to make sure that it is stored and used responsibly. Everyone should be able to find out about how patient data are used. #datasaveslives You can find out more about the background to this citation here: https://understandingpatientdata.org.uk/data-citation.
Disclaimers
This report presents independent research funded by the National Institute for Health Research (NIHR). The views and opinions expressed by authors in this publication are those of the authors and do not necessarily reflect those of the NHS, the NIHR, CCF, NETSCC, PGfAR or the Department of Health and Social Care. If there are verbatim quotations included in this publication the views and opinions expressed by the interviewees are those of the interviewees and do not necessarily reflect those of the authors, those of the NHS, the NIHR, NETSCC, the PGfAR programme or the Department of Health and Social Care.