Feasibility study for supporting medication adherence for adults with cystic fibrosis: mixed-methods process evaluation

Hind, Daniel; Drabble, Sarah J; Arden, Madelynne A; Mandefield, Laura; Waterhouse, Simon; Maguire, Chin; Cantrill, Hannah; Robinson, Louisa; Beever, Daniel; Aj, Scott; Keating, Samuel; Hutchings, Marlene; Bradley, Judy; Nightingale, Julia A.; Allenby, Mark I.; Dewar, J.; Whelan, Pauline; Ainsworth, John; Walters, Stephen J.; Wildman, Martin J; O’Cathain, Alicia

doi:10.1136/bmjopen-2020-039089

Cited by 9 publications

(7 citation statements)

References 98 publications

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“…The feasibility study led to 25 key changes to RCT procedures and the intervention. 43 Plans to develop an app for mobile phones were progressed. Changes were made to CFHealthHub to make it easier for interventionists to view and edit prescription data and to handle alternating treatment regimens.…”

Section: Resultsmentioning

confidence: 99%

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An intervention to support adherence to inhaled medication in adults with cystic fibrosis: the ACtiF research programme including RCT

Wildman

O’Cathain

Hind

et al. 2021

Programme Grants Appl Res

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Background People with cystic fibrosis frequently have low levels of adherence to inhaled medications. Objectives The objectives were to develop and evaluate an intervention for adults with cystic fibrosis to improve adherence to their inhaled medication. Design We used agile software methods to develop an online platform. We used mixed methods to develop a behaviour change intervention for delivery by an interventionist. These were integrated to become the CFHealthHub intervention. We undertook a feasibility study consisting of a pilot randomised controlled trial and process evaluation in two cystic fibrosis centres. We evaluated the intervention using an open-label, parallel-group randomised controlled trial with usual care as the control. Participants were randomised in a 1 : 1 ratio to intervention or usual care. Usual care consisted of clinic visits every 3 months. We undertook a process evaluation alongside the randomised controlled trial, including a fidelity study, a qualitative interview study and a mediation analysis. We undertook a health economic analysis using both a within-trial and model-based analysis. Setting The randomised controlled trial took place in 19 UK cystic fibrosis centres. Participants Participants were people aged ≥ 16 years with cystic fibrosis, on the cystic fibrosis registry, not post lung transplant or on the active transplant list, who were able to consent and not using dry-powder inhalers. Intervention People with cystic fibrosis used a nebuliser with electronic monitoring capabilities. This transferred data automatically to a digital platform. People with cystic fibrosis and clinicians could monitor adherence using these data, including through a mobile application (app). CFHealthHub displayed graphs of adherence data as well as educational and problem-solving information. A trained interventionist helped people with cystic fibrosis to address their adherence. Main outcome measures Randomised controlled trial – adjusted incidence rate ratio of pulmonary exacerbations meeting the modified Fuchs criteria over a 12-month follow-up period (primary outcome); change in percentage adherence; and per cent predicted forced expiratory volume in 1 second (key secondary outcomes). Process evaluation – percentage fidelity to intervention delivery, and participant and interventionist perceptions of the intervention. Economic modelling – incremental cost per quality-adjusted life-year gained. Results Randomised controlled trial – 608 participants were randomised to the intervention (n = 305) or usual care (n = 303). To our knowledge, this was the largest randomised controlled trial in cystic fibrosis undertaken in the UK. The adjusted rate of exacerbations per year (primary outcome) was 1.63 in the intervention and 1.77 in the usual-care arm (incidence rate ratio 0.96, 95% confidence interval 0.83 to 1.12; p = 0.638) after adjustment for covariates. The adjusted difference in mean weekly normative adherence was 9.5% (95% confidence interval 8.6% to 10.4%) across 1 year, favouring the intervention. Adjusted mean difference in forced expiratory volume in 1 second (per cent) predicted at 12 months was 1.4% (95% confidence interval –0.2% to 3.0%). No adverse events were related to the intervention. Process evaluation – fidelity of intervention delivery was high, the intervention was acceptable to people with cystic fibrosis, participants engaged with the intervention [287/305 (94%) attended the first intervention visit], expected mechanisms of action were identified and contextual factors varied between randomised controlled trial sites. Qualitative interviews with 22 people with cystic fibrosis and 26 interventionists identified that people with cystic fibrosis welcomed the objective adherence data as proof of actions to self and others, and valued the relationship that they built with the interventionists. Economic modelling – the within-trial analysis suggests that the intervention generated 0.01 additional quality-adjusted life-years at an additional cost of £865.91 per patient, leading to an incremental cost-effectiveness ratio of £71,136 per quality-adjusted life-year gained. This should be interpreted with caution owing to the short time horizon. The health economic model suggests that the intervention is expected to generate 0.17 additional quality-adjusted life-years and cost savings of £1790 over a lifetime (70-year) horizon; hence, the intervention is expected to dominate usual care. Assuming a willingness-to-pay threshold of £20,000 per quality-adjusted life-year gained, the probability that the intervention generates more net benefit than usual care is 0.89. The model results are dependent on assumptions regarding the duration over which costs and effects of the intervention apply, the impact of the intervention on forced expiratory volume in 1 second (per cent) predicted and the relationship between increased adherence and drug-prescribing levels. Limitations Number of exacerbations is a sensitive and valid measure of clinical change used in many trials. However, data collection of this outcome in this context was challenging and could have been subject to bias. It was not possible to measure baseline adherence accurately. It was not possible to quantify the impact of the intervention on the number of packs of medicines prescribed. Conclusions We developed a feasible and acceptable intervention that was delivered to fidelity in the randomised controlled trial. We observed no statistically significant difference in the primary outcome of exacerbation rates over 12 months. We observed an increase in normative adherence levels in a disease where adherence levels are low. The magnitude of the increase in adherence may not have been large enough to affect exacerbations. Future work Given the non-significant difference in the primary outcome, further research is required to explore why an increase in objective normative adherence did not reduce exacerbations and to develop interventions that reduce exacerbations. Trial registration Work package 3.1: Current Controlled Trials ISRCTN13076797. Work packages 3.2 and 3.3: Current Controlled Trials ISRCTN55504164. Funding This project was funded by the National Institute for Health Research (NIHR) Programme Grants for Applied Research programme and will be published in full in Programme Grants for Applied Research; Vol. 9, No. 11. See the NIHR Journals Library website for further project information.

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Section: Resultsmentioning

confidence: 99%

“…This was a pilot, open-label, parallel-group RCT with concurrent mixed-methods process evaluation. The pilot RCT has been reported in Hind et al 41 The qualitative research from the process evaluation has been reported in Drabble et al 42 and Hind et al 43…”

Section: Designmentioning

confidence: 99%

An intervention to support adherence to inhaled medication in adults with cystic fibrosis: the ACtiF research programme including RCT

Wildman

O’Cathain

Hind

et al. 2021

Programme Grants Appl Res

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“…The sample size of this study was limited by budget and time constraints, so we may not have reached saturation in barriers and solutions sampled. While participants using CFHealthHub generally welcomed the video library, some preferred not to view videos to avoid comparison with people like them who have more severe CF or people like them who seemed healthier [ 15 ]. Although all participants in the RCT intervention arm had access to the video library, we emphasised that video viewing was optional in recognition that some people with CF prefer to avoid social comparison [ 16 ].…”

Section: Discussionmentioning

confidence: 99%

Modelling Successful Self-Management in Adults With Cystic Fibrosis: Vicarious Self-Efficacy From Videos of ‘People Like Me’

Hutchings¹,

Kirkpatrick²,

Arden³

et al. 2022

Cureus

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BackgroundSelf-efficacy is an important determinant of treatment adherence, and peer modelling of success can provide vicarious self-efficacy. A series of patient stories ('talking heads' videos) were developed with people with cystic fibrosis (CF) as part of the CFHealthHub multi-component adherence intervention, aiming to demonstrate success with daily therapy in 'people like me'. MethodologyOne-to-one semi-structured interviews exploring patients' experiences, barriers and facilitators of nebuliser adherence were audio and video-recorded between October 2015 and August 2016. Interview transcripts were reviewed to identify descriptions of problem-solving and sustained treatment success. Positive stories potentially providing vicarious descriptions of success were selected as video clips. ResultsIn total, 14 adults with CF were recruited from five UK CF centres. Each participant contributed a median of five (interquartile range: 3-6) video clips, and a total of 57 unique clips were uploaded onto the CFHealthHub digital platform. Nine of those clips spanned two categories, hence, there were 66 clips across 16 categories. ConclusionsThe videos were well received though some adults were concerned that comparisons with peers might create anxiety by highlighting the possibility of future decline or current relative underperformance. It is important to sensitively support choice when providing resources aiming to increase vicarious self-efficacy. Our experience may guide the development of similar videos for people with other long-term conditions.

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“…immediately following the delivery. [175][176][177] Qualitative interviews of delivery agents can also be conducted to gauge views regarding aspects of an intervention such as the training received to deliver, con dence in delivering, and any perceived barriers to delivering an intervention as planned. 172 Factors affecting delity can be collected to understand what, if anything, may in uence departures from delivering an intervention as designed.…”

Section: Fidelitymentioning

confidence: 99%

Expert Perspectives on Pilot and Feasibility Studies: A Delphi Study and Consolidation of Considerations for Behavioral Interventions

Pfledderer,

von Klinggraeff,

Burkart

et al. 2023

Preprint

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Background In the behavioral sciences, conducting pilot and/or feasibility studies (PFS) is a key step that provides essential information used to inform the design, conduct, and implementation of a larger-scale trial. There are more than 160 published guidelines, reporting checklists, frameworks, and recommendations related to PFS. All of these publications offer some form of guidance on PFS, but many focus on one or a few topics. This makes it difficult for researchers wanting to gain a broader understanding of all the relevant and important aspects of PFS and requires them to seek out multiple sources of information, which increases the risk of missing key considerations to incorporate into their PFS. The purpose of this study was to develop a consolidated set of considerations for the design, conduct, implementation, and reporting of PFS for interventions conducted in the behavioral sciences. Methods To develop this consolidation, we undertook a review of the published guidance on PFS in combination with expert consensus (via a Delphi study) from the authors who wrote such guidance to inform the identified considerations. A total of 161 PFS-related guidelines, checklists, frameworks, and recommendations were identified via a review of recently published behavioral intervention PFS and backward/forward citation tracking of well-know PFS literature (e.g., CONSORT Ext. for PFS). Authors of all 161 PFS publications were invited to complete a three-round Delphi survey, which was used to guide the creation of a consolidated list of considerations to guide the design, conduct, and reporting of PFS conducted by researchers in the behavioral sciences. Results A total of 496 authors were invited to take part in the Delphi survey, 50 (10.1%) of which completed all three rounds, representing 60 (37.3%) of the 161 identified PFS-related guidelines, checklists, frameworks, and recommendations. A set of twenty considerations, broadly categorized into six themes (Intervention Design, Study Design, Conduct of Trial, Implementation of Intervention, Statistical Analysis and Reporting) were generated from a review of the 161 PFS-related publications as well as a synthesis of feedback from the three-round Delphi process. These 20 considerations are presented alongside a supporting narrative for each consideration as well as a crosswalk of all 161 publications aligned with each consideration for further reading. Conclusion We leveraged expert opinion from researchers who have published PFS-related guidelines, checklists, frameworks, and recommendations on a wide range of topics and distilled this knowledge into a valuable and universal resource for researchers conducting PFS. Researchers may use these considerations alongside the previously published literature to guide decisions about all aspects of PFS, with the hope of creating and disseminating interventions with broad public health impact.

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Feasibility study for supporting medication adherence for adults with cystic fibrosis: mixed-methods process evaluation

Cited by 9 publications

References 98 publications

An intervention to support adherence to inhaled medication in adults with cystic fibrosis: the ACtiF research programme including RCT

An intervention to support adherence to inhaled medication in adults with cystic fibrosis: the ACtiF research programme including RCT

Modelling Successful Self-Management in Adults With Cystic Fibrosis: Vicarious Self-Efficacy From Videos of ‘People Like Me’

Expert Perspectives on Pilot and Feasibility Studies: A Delphi Study and Consolidation of Considerations for Behavioral Interventions

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