A systematic review to determine the presence and effectiveness of shared decision making interventions for airway clearance techniques in adults with bronchiectasis

Introduction

Bronchiectasis is a chronic lung disorder associated with poor quality of life and frequent exacerbations (Polverino et al. 2017). It is characterised radiologically by permanent dilation of the bronchi, and clinically by a combination of physical symptoms including cough, sputum production and recurrent respiratory infections (Chalmers & Hill 2013).

People with bronchiectasis experience chronic productive cough and acute exacerbations, which are linked to poorer quality of life and a higher rate of disease progression (Lee et al. 2021). Higher disease progression carries an increased risk of hospitalisation (Chalmers et al. 2014; Costa et al. 2018) where currently over £30 million is spent per year in the U.K. (Goeminne et al. 2019). There is an estimated 25% mortality rate for patients with severe disease within 4 years (Menéndez et al. 2017).

Impaired muco-ciliary clearance and sputum retention are core elements in the pathophysiology of bronchiectasis. Consensus guidelines recommend that all patients with bronchiectasis receive airway clearance techniques (ACTs) (Polverino et al. 2017). Despite these recommendations, reported use of ACTs vary significantly throughout the world. Data from the European Bronchiectasis Registry (n = 13,512) show only around 50% of patients perform regular ACTs, ranging from 10% in Sweden to 92% in Denmark (Spinou et al. 2020); whereas an analysis from the United States by Basavaraj et al. (2020) showed slightly higher average reported use of daily ACTs, 59% (n = 905).

Airway clearance techniques

ACTs are non-pharmacological interventions that facilitate removal of secretions from the lungs (Bradley et al. 2018). A myriad of ACTs are applied in clinical practice, including positioning, gravity-assisted drainage, manual techniques, various breathing strategies, positive expiratory pressure (PEP) devices, oscillating positive expiratory (OPEP) devices and mechanical tools that are applied to the external chest wall (Lee et al. 2017). Many of these ACTs may be used in isolation or in combination with one another.

See Figure 1: Most commonly used airway clearance technique in each country (Spinou et al. 2020)

PEP = positive expiratory pressure; AECOPD = acute exacerbation of chronic obstructive pulmonary disease; ACBT = active cycle of breathing technique; MTs = manual techniques; FET = forced expiratory technique; CF = cystic fibrosis.

The current prescription of ACTs by respiratory physiotherapists and other appropriate health care professionals (HCPs) varies globally. Figure 1 shows the most commonly prescribed ACTs across the world. Factors influencing regional trends in ACT are complex including clinician familiarity and training, reimbursement approvals particular to each healthcare system, clinical care pathways and patient preferences (Hoo et al. 2015). For example, an online survey conducted by the Association of Chartered Physiotherapists in Respiratory Care (ACPRC) found that 44% of U.K. physiotherapists (n = 63) struggled with funding of PEP/OPEP devices in their respective healthcare environment which affected their decision on the type of ACT they could prescribe (ACPRC 2020).

Guidance of ACT prescription has emerged in the past two years with the publication of the British Thoracic Society (BTS) guidelines for bronchiectasis in adults (Hill et al. 2019a). These guidelines include a flow chart recommending which ACTs to prescribe and when (for example, patients in a stable state and those with acute inpatient exacerbation), Figures 2 and 3 respectively. The availability of this flow chart may be viewed as both a positive and negative step in ACT prescription. It provides clear instructions to respiratory physiotherapists on initial ACT prescription (ACBT +/- postural drainage) and considerations of adjuncts when this is not effective (for example, OPEP). Alternatively, the flow chart could be simply followed to the letter, with little or no consideration for a more personalised approach as previously mentioned, including patient preference or adherence.

See Figure 2: Physiotherapy management – stepwise airway clearance techniques during a stable state. Adapted from The British Thoracic Society guideline for bronchiectasis in adults (Hill et al. 2019)

ACBT = active cycle of breathing techniques.

See Figure 3: Physiotherapy management – stepwise airway clearance techniques during an exacerbation. Adapted from The British Thoracic Society guideline for bronchiectasis in adults (Hill et al. 2019)

PD = postural drainage; mPD = modified postural drainage.

Shared decision making as an intervention

SDM is an approach where clinicians and patients are expected to make decisions together, using the best available evidence (Elwyn et al. 2010b). Patients and service users should be able to understand the care, treatment and support options available to them, including the benefits and risks associated with those options (NHS England 2019).

SDM interventions such as ‘decision aids’ have already been designed for a range of clinical specialities including cancer and diabetes (Elwyn et al. 2010a; Trikalinos et al. 2015). As there is only low-grade evidence for ACTs in bronchiectasis and little evidence that one technique is superior to others; SDM may be a feasible intervention to improve patient choice of, and adherence to ACT’s.

Interventions may include but are not limited to; option grids during consultations listing the range of ACTs available based on current evidence and the pros and cons of each; paper or electronic based decision aids with comprehensive or up to date and evidenced based information on all types of ACTs that patients can bring home and independently decide on what type of, if any, ACT they wish to use. Additionally, specific behavioural change techniques (BCTs) for example, behavioural regulation, beliefs about benefits and motivation, could be used to facilitate SDM within the consultation.

Why is it important to do this review?

There is currently a lack of RCTs proving the efficacy of one specific airway clearance technique over another in bronchiectasis (Hill et al. 2019b). SDM tools are usually designed for situations where there is uncertainty about the best treatment option and provide information about the advantages and disadvantages in as balanced a way as possible (Elwyn et al. 2010b); lending them well to a patient preference situation where the clinician is in clinical equipoise.

The integration of SDM in clinical practice can help indicate to the patient that their opinions and preferences are valued and that patient-centred care has been achieved (Carmona et al. 2021). National bronchiectasis guidelines state that patient preference and adherence should be considered when recommending ACTs (Hill et al. 2019a) but provides no indication on how this should be performed.

This systematic review seeks to establish if SDM is used when choosing ACTs for adult patients with bronchiectasis and if possible, determine the effectiveness of this intervention. The review aims to identify, appraise and summarise the literature from which a specific SDM framework could be established or a decision tool developed, trialled and adopted in national guidelines.

Aims

This systematic review aims:

• To determine if and how SDM is used when choosing ACTs for adults with bronchiectasis.

• To determine the effectiveness of SDM when choosing ACTs for adults with bronchiectasis.

Effectiveness in this review will be measured using clinical and patient outcomes. Clinical outcomes will include: exacerbation frequency, hospitalisation, adverse events and mortality. Patient outcomes will include: patient adherence, health related quality of life, patient preference and acceptance.

Methods

The protocol was registered on the international Prospective Register of Systematic Reviews (PROSPERO) database on 17th June 2021 (registration number: CRD42021261640). We have conformed to the Preferred reporting items for systematic reviews and meta-analyses (PRISMA) (Moher et al. 2010) herein.

Eligibility criteria

The following inclusion and exclusion criteria were used to guide the screening and selection of studies in the systematic review.

Inclusion criteria

• Adults ≥18. Confirmed clinical and radiological diagnosis of bronchiectasis. Co-morbid respiratory disease such as asthma and COPD will be included.

• Any intervention using shared decision making for example, one-to-one basis, a group basis, discussion sessions, role play sessions, blended learning sessions, online learning sessions and the use of hard-copy information resources such as leaflets or workbooks or option grids. This includes all interventions named as promoting, improving, enabling or facilitating shared decision making.

• The use of any ACTs by patients.

• Presence of shared decision-making measured by any validated tool including but not limited to:

• The Observing patient involvement 12-item (OPTION) scale (Elwyn et al. 2003).

• The Observer-based measure observer 5-item (OPTION) scale (Elwyn et al. 2013).

• Decision-making instrument facilitation antecedents (for example, the Preparation for decision-making scale) (Bennett et al. 2010).

• Decision process (for example, the Rochester participatory decision-making scale) (Shields et al. 2005).

• Clinical outcomes including: exacerbation frequency, lung function, hospitalisation, sputum characteristics, adverse events and mortality.

• Patient outcomes including: patient adherence, health related quality of life, patient satisfaction, decision regret and patient preference and acceptance.

• All study types except case reports, expert opinion and editorials will be included.

Exclusion criteria

• Children <18 years old.

• CF as a co-morbidity.

• Other isolated respiratory diseases for example, Asthma, COPD, CF.

• Non-English publications.

Publication date

No limit was set for publication date. These varied between databases.

Search criteria

The search strategy was developed by PM with support from FB, and then piloted on 28th June 2021 to ensure it was comprehensive enough to identify as many appropriate studies as possible. The electronic searches took place between June and July 2021.

Electronic searches

A systematic literature review was conducted using the following electronic databases: CINAHL, EMBASE, Medline, PsycINFO, Google Scholar, Web of Science and the Cochrane Library (Cochrane Database of Systematic Reviews, Cochrane Central Register of Controlled Trials, Health Technology Assessments Database, Cochrane Airways Group).

Additionally a search of the clinical trials registries, ClinicalTrials.gov (www.ClinicalTrials.gov) and grey literature through Open Grey (www.opengrey.eu) and grey matters (www.cadth.ca/grey-matters-practical-tool-searching-health-related-grey-literature-0) was completed. All databases were searched from their inception to 29th June 2021. Due to time and financial constraints, a restriction on non-English publications was imposed. A report on any eligible non-English publications will be made, specifically stating any evidence of potential language bias in the review.

Additional searches

The online Medical Decision-Making journal was also searched using the terms ‘bronchiectasis’ and ‘airway clearance’ on 22nd July 2021.

Search terms

Search terms were developed from the review questions which was derived from the PICO (Population, Intervention, Comparison, Outcome) and PEO (Population, Exposure, Outcome) frameworks (Schardt et al. 2007; Bettany-Saltikov 2016). Table 1 illustrates the key PICO and PEO search terms.

Table 1: ‘PICO’ and ‘PEO’ keyword search terms.

Population	Intervention	Exposure	Comparison	Outcome
Adults with bronchiectasis	Shared decision making	Airway clearance*	Usual care	Exacerbation frequency
Adults with non-cystic fibrosis bronchiectasis	Attitude of health personnel	Mucus clearance*		Adherence
Bronchiectasis	Attitude to health	Sputum clearance*		St. Georges quality of life questionnaire
Non-cystic fibrosis bronchiectasis	Choice behavio*	Secretion clearance*		Patient acceptance
	Communication	Active cycle of breathing*		Patient satisfaction
	Decision support technique*	Positive expiratory pressure*		Lung function
	Decision making	Manual technique*		Hospitalisation

* = truncation of terms.

Study selection

All search results including title, author(s) and abstract fields were downloaded and imported to EndNote X9. EndNote was used to identify and remove all duplicates. Once all duplicates were removed all articles were imported to ‘Rayyan’. Rayyan is an online platform which allows researchers to conduct initial screening of abstracts and titles for systematic reviews (Ouzzani et al. 2016). All studies uploaded to Rayyan were screened using a template derived from the eligibility criteria of the review.

Data extraction

All data was extracted into pre-defined data extraction form. The data extraction form (Appendix 1) was designed specifically for this review. The data extraction form includes participant demographics, aims and methods of the study, data and author findings and quality assessment.

Quality assessment of included studies

This review used Critical Appraisal Skills Programme (CASP) checklists (Appendix 2). The CASP tool is the most commonly used tool for quality appraisal in health-related qualitative evidence syntheses, with endorsement from the Cochrane Qualitative and Implementation Methods Group (Long et al. 2020).

Data analysis and synthesis

We planned to use tables with supporting narrative to determine whether the included studies were sufficiently similar in design, participants, interventions and outcomes to be combined in a meta-analysis (Schünemann et al. 2008). We intended to use a random-effects model using standardised mean differences with a 95% confidence interval. Standardised mean difference, with 95% confidence intervals would have been used where outcome measures such as lung function or health related quality of life are the same, but interventions varied in either methods or outcome measure scales. If appropriate, we planned to use forest plots to assess heterogeneity using i2.

To ensure robustness of any summary statistics, we planned to perform sensitivity analysis if there were sufficient comparable studies. This would have involved adding or removing studies where there was high risk of bias in relation to randomisation, allocation concealment, or blinding of the interventions from participants or trial personnel (Deeks et al. 2011).

Due to the anticipated heterogeneity in study design, methods and methodology; a narrative synthesis was planned for the review synthesis. This narrative synthesis used the explicit framework proposed by Popay et al. (2006); developing a theory of change model; developing a preliminary synthesis, exploring relationships within and between studies, and assessing the robustness of the synthesis.

Results

A total of 4414 studies were initially identified from the searches. 864 duplicate studies were removed; resulting in 3550 studies in total for title and abstract screening. All 3550 titles and abstracts underwent double-blind screening by the lead reviewer (PM) and co-authors (JB, SM, JB, ADS). Disagreements for inclusion or exclusion were resolved through discussion.

Seventeen studies were eligible for full text screening. Two studies (Lawton et al. 2019; Ryan & MacLeod, 2020) were removed as no full text were available. The remaining full text studies (n = 15) were screened independently by two reviews, PM and ADS. Any uncertainties were resolved though discussion. A third reviewer (SM) was used as mediator where consensus for studies was not reached.

All references of the 15 full text studies were searched manually by the lead researcher and compared to the list created in EndNote to ensure there were no missing data. Where studies were found that were not in the EndNote library; they were screened with the proforma used in the title and abstract screening in this review. No additional relevant studies were identified.

Description of results

No studies were identified for inclusion in the review. We have presented a study flow diagram illustrating the results (Figure 4) and reasons for exclusion (Table 2).

See Figure 4: Preferred reporting items for systematic reviews and meta-analysis (PRISMA) flow diagram.

Table 2: Reasons for exclusion

Study	Reason for exclusion
Brockwell et al. (2020)	Not an appropriate intervention. Mentions ‘Bronchiectasis Education Tool’ as an intervention. States ‘decision making’ was not taught as part of the intervention
Cecins et al. (1999)	Not an appropriate intervention. Includes COPD patients as well as bronchiectasis
Chalmers et al. (2014)	Not appropriate study design (review article)
Eaton et al. (2007)	Not an appropriate intervention. Various techniques, no SDM but patient preference measured
Farley et al. (2008)	Not appropriate study design (review article)
Flude et al. (2012)	Not appropriate study design (review article)
Guan et al. (2019)	Not an appropriate intervention. Does not mention SDM specific to ACT, only ‘self-management techniques’
Kelly et al. (2018)	Not an appropriate intervention. Includes SDM in references but not as focus of work
Kelly et al. (2021)	Not an appropriate intervention. Mentions SDM only in discussion ‘making autonomous decision under direction of clinician’ in relation to ACTs
Knowles et al. (2021)	Not appropriate study design (review article). In discussion the author mentions ‘respiratory physiotherapy should ensure there is SDM regarding patients’ preferences’
Lavery et al. (2007)	Not an appropriate intervention. Does not mention SDM
Lavery et al. (2011)	Not an appropriate intervention. Does not mention SDM
Lawton et al. (2019)	No full text available (abstract only)
Lee et al. (2021)	Not an appropriate intervention. Study mentions a mix of ACTs were prescribed which ‘aligns’ with acceptable methods such as considering patient preference. Does not explicitly state this study used SDM when prescribing ACTs
McIlwaine et al. (2017)	Not appropriate study design (review article)
Herrero-Cortina et al. (2016)	Not an appropriate intervention. Study measures an element of SDM (patient preference) as an outcome, not as part of the intervention
Ryan and MacLeod (2020)	No full text available (abstract only)

Discussion

Patient involvement in decision-making is becoming an essential element of modern medicine. Recent healthcare policy-making and legislation provide guidance on why SDM should be part of everyday care in all healthcare setting (NHS England 2019; Carmona et al. 2021; National Institute for Health and Care Excellence 2021). This systematic review provides a timely contribution by demonstrating the gap in evidence of this in airway clearance techniques for adults with bronchiectasis.

This review identified the lack of evidence that SDM is used when choosing airway clearance techniques for adults with bronchiectasis. The review subsequently concludes insufficient evidence to demonstrate the effectiveness of SDM interventions for choosing airway clearance techniques in adults with bronchiectasis.

Brockwell et al. (2020) found that improved interaction and communication with healthcare professionals on self-management techniques including airway clearance was the primary theme in their analysis of patient focus groups using the Bronchiectasis Education Tool (BET). They concluded patients have a desire to be involved with and assist initiatives to increase their education of ACTs to support their condition.

Similarly in a study by Kelly et al. (2021) exploring views of self-management with respiratory physiotherapists and adult patients with bronchiectasis they found making autonomous decisions under the direction and support of a clinician was recognised as a significant part of self-management by patients. When Kelly et al. (2021) specifically looked at patient influencers on self-management, they concluded there is a need for tools to promote participation in education on ACTs that are acceptable to patients and do not add to their treatment burden.

An unpleasantly familiar, frequently published, yet unchanging statistic over the past 20 years, is the low adherence rates (averaging 30%) of ACTs within many respiratory diseases including bronchiectasis, CF and COPD (White et al. 2007; Flores et al. 2013; Bradley et al. 2018; Low et al. 2020). A recent systematic review into barriers and facilitators for ACTs in bronchiectasis found a lack of time the most common reason for not performing them, with other reasons such as competing priorities and lack of perceived benefit from adherence also frequently cited (Low et al. 2020). Given, longer-term adherence is essential to identifying long-term clinical benefit for ACTs in bronchiectasis; it may be pragmatic to consider some trade-off on efficacy if patients were able to make an informed choice. An informed choice will only be possible if there is a more personalised approach to the prescription of ACTs in bronchiectasis such as SDM.

Limitations of this review

Due to resource limitation, this review was limited to English language publications only. However, throughout title and abstract screening, the authors did not identify any study that met all the inclusion criteria except publication in English.

With no studies meeting criteria for inclusion, a limitation of this review may be that it appears to offer no conclusions or offer conclusions not based on evidence and may seem disappointing among some clinicians. We argue that this empty review remains important and highlights a major research gap and has identified the state of the evidence at this point in time in SDM for ACTs in bronchiectasis.

Implications for practice

No eligible studies were found for inclusion in this review. We would argue this has serious implications for practice. We were unable to identify the use or effectiveness of any SDM intervention in airway clearance techniques in adults with bronchiectasis. Based on the literature examined during this systematic review, there appears to be a desire from both patients and health professionals to engage with elements of SDM to facilitate a personalised ACT prescription that takes into account the patient’s disease state, preference and motivation, together with the physiological knowledge base of each ACT (Flude et al. 2012; Herrero-Cortina et al. 2016; McIlwaine et al. 2017; Hester et al. 2018; Kelly et al. 2018; Knowles et al. 2021). The author acknowledges that this desire and ability to participate in SDM, may differ significantly between patients who have been recruited into research studies to allow such conclusions, and those who have not. This selection or recruitment bias may not reflect a ‘real world’ demand for and engagement of a potential SDM intervention.

Implications for research

A lack of studies for inclusion in this review has identified a gap in research focusing on SDM interventions for ACTs in adults with bronchiectasis. We hope that having identified this gap in research, we have created a need to design high-quality SDM interventions for ACTs in adults with bronchiectasis amongst clinicians and researchers.

Qualitative studies may play an important role in the development of SDM interventions for ACTs in adults with bronchiectasis. For example, studies looking at patient preferences in the delivery of information, format of the intervention for example, electronic/hard copy decision aids, BCTs or a combination of these, may lay the foundations for identification of interventions that could be tested in feasibility trials up to high-quality RCTs.

Conclusion

Bronchiectasis is an increasingly prevalent disease. ACTs are the cornerstone of bronchiectasis management. We have presented clear justification for further research for development of a SDM intervention for ACTs in adults with bronchiectasis. This is supported by the recent NICE guideline on SDM, which made specific recommendations for research including: research on differing SDM interventions in different groups and the acceptability of these SDM interventions (National Institute for Health and Care Excellence 2021). We hope to see progress in this field in the near future to assess any impact it may have for this population.

Key points

1 There is a gap in research focusing on SDM interventions for ACTs in adults with bronchiectasis.

2 Patients and healthcare professionals have an enthusiasm to engage with and promote SDM in airway clearance techniques respectively.

3 There is a justification for further research for development of SDM interventions for ACTs in adults with bronchiectasis.

Statement of ethics

An ethics statement is not applicable because this study is based exclusively on published literature.

Conflict of interest statement

None.

Funding sources

This research was funded by the National Institute of Health Research (NIHR) during the corresponding authors HEE/NIHR ICA Programme Pre-doctoral Clinical Academic Fellowship (award reference: NIHR300310).

Acknowledgements

The lead author would like to thank F. Beyer for her contributions for this piece of work.

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