Purpose of the PSP and background
The purpose of this protocol is to clearly set out the aims, objectives and commitments of the Breathlessness Priority Setting Partnership (PSP) in line with James Lind Alliance (JLA) principles. The Protocol is a JLA requirement and will be published on the PSP’s page of the JLA website. The Steering Group will review the Protocol regularly and any updated version will be sent to the JLA.
The JLA is a non-profit making initiative, established in 2004. It brings patients, carers and clinicians together in PSPs. These PSPs identify and prioritise the evidence uncertainties, or ‘unanswered questions’, that they agree are the most important for research in their topic area. Traditionally PSPs have focused on uncertainties about the effects of treatments, but some PSPs have chosen to broaden their scope beyond that. The aim of a PSP is to help ensure that those who fund health research are aware of what really matters to patients, carers and clinicians. The National Institute for Health and Care Research (NIHR) coordinates the infrastructure of the JLA to oversee the processes for PSPs, based at the NIHR Coordinating Centre (NIHRCC), University of Southampton.
Breathlessness is the most prevalent symptom across respiratory conditions although it can also occur as result of other conditions. Approximately two thirds of cases of breathlessness in adults are due to a respiratory or cardiac disorder. It is more prevalent than cough in advanced respiratory diseases and is usually more severe. It can be difficult to articulate but is terrifying to experience. It is a major cause of suffering in people with respiratory diseases, however it also transcends disease categories and we therefore propose to limit the scope of this project to breathlessness as a symptom, irrespective of the underlying condition(s) which may be causing it.
Breathlessness reduces life expectancy and increases the chances of experiencing unemployment, sickness absence and lower productivity. A staggering £1.2 billion is lost annually in the UK due to work days lost. Breathlessness becomes more severe as the disease progresses, contributing to one in five emergency department attendances by ambulance. Breathless patients are more likely to present “out-of-hours” and are twice as likely to be admitted to hospital compared with those attending for other reasons. Addressing breathlessness effectively can slow or prevent the progression of disease while leading to benefits for the individual and the wider society.
Asthma + Lung UK would like to see an increased focus (and increased investment) on research into breathlessness, and establishing research and innovation priorities is essential to ensuring funding is focussed on the most important challenges that, if solved, will lead to the biggest benefit for patients.
Aims, objectives and scope of the PSP
The aim of the breathlessness PSP is to identify the unanswered questions from patient, carer and clinical perspectives and then prioritise those that patients, carers and clinicians agree are the most important for research to address.
The objectives of the PSP are to:
- work with patients, carers and clinicians to identify uncertainties about the diagnosis, treatment, self-management and prevention of breathlessness
- to agree by consensus a prioritised list of those uncertainties, for research
- to publicise the results of the PSP and process
- to take the results to research commissioning bodies to be considered for funding.
The scope of the breathlessness PSP is defined as:
- breathlessness as a symptom, including diagnosis, treatment and side effects, but irrespective of the specific underlying condition(s) which may be causing it.
The PSP will exclude from its scope questions about:
- The underlying condition(s) which is/are leading to breathlessness
- Breathlessness in babies and infants (<2years)
- Individual experience of provision and or quality of healthcare and services
- Non-UK perspectives
The Steering Group is responsible for discussing what implications the scope of the PSP will have for the evidence-checking stage of the process. Resources and expertise will be put in place to do this evidence checking.
The Steering Group
The Steering Group includes membership of patients and carers and clinicians , as individuals or representatives from a relevant group.
The breathlessness PSP will be led and managed by a Steering Group involving the following:
Patient and carer representative/s
Donna Key
David Burnley
Megan Lewis
Melissa Holston
Clinical representative/s
Dr. Rachael Evans, Clinical Associate Professor and Honorary Consultant Respiratory Physician, University of Leicester
Dr. Charlie Reilly, Consultant Physiotherapist, Kings College London
Beverley Bostock, Advanced Nurse Practitioner, Mann Cottage Surgery
Dr. Kyle Pattinson, Consultant Anaesthetist and Associate Professor, Nuffield Department of Clinical Neurosciences
Dr. Katherine Hickman, GP and Respiratory lead, West Yorkshire ICB,
Prof Jaclyn Smith, Consultant, Manchester University NHS Foundation Trust
Project coordinator
Dr. Erika Kennington, Asthma + Lung UK: JLA Project Lead
Heather Thorn, Asthma + Lung UK: JLA Project coordinator
Suzannah Kinsella, James Lind Alliance: James Lind Alliance Adviser and Chair of the Steering Group
Dr. Hannah Springs: Information Specialist
The Steering Group will agree the resources, including time and expertise that they will be able to contribute to each stage of the process, with input and advice from the JLA.
Partners
Organisations and individuals will be invited to be involved with the PSP as partners. Partners are individuals, organisations or groups who will commit to supporting the PSP, promoting the process and encouraging their represented groups or members to participate. Organisations which can reach and advocate for these groups will be invited to become involved in the PSP. Partners represent the following groups:
- people who have had, or have, breathlessness secondary to a lung condition:
- Asthma + Lung UK
- Cystic Fibrosis Trust
- Action for Pulmonary Fibrosis
- Pulmonary Fibrosis Trust
- Mesothelioma UK
- Roy Castle Foundation
- Long Covid support groups
- carers of people who have had, or have, breathlessness secondary to a lung condition
- health and social care professionals - with experience of breathless secondary to a lung condition:
- The Dyspnoea Society
- Primary Care Respiratory Society
- Complex Breathlessness group
- Association of Respiratory Nurses
- Primary Care Cardiovascular Society
- King’s College Hospital NHS Foundation Trust
- Cicley Saunders International
Exclusion criteria
Some organisations may be judged by the JLA or the Steering Group to have conflicts of interest. These may be perceived to potentially cause unacceptable bias as a member of the Steering Group. As this is likely to affect the ultimate findings of the PSP, those organisations will not be invited to participate. It is possible, however, that interested parties may participate in a purely observational capacity when the Steering Group considers it may be helpful.
The methods the PSP will use
This section describes a schedule of proposed steps through which the PSP aims to meet its objectives. The process is iterative and dependent on the active participation and contribution of different groups. The methods used in any step will be agreed through consultation between the Steering Group members, guided by the PSP’s aims and objectives. More details of the method are in the Guidebook section of the JLA website at where examples of the work of other JLA PSPs can be seen.
Step 1: Identification and invitation of potential partners
Potential partner organisations will be identified through a process of peer knowledge and consultation, through the Steering Group members’ networks. Potential partners will be contacted and informed of the establishment and aims of the breathlessness PSP.
Step 2: Awareness raising
PSPs will need to raise awareness of their proposed activity among their patient, carer and clinician communities, in order to secure support and participation. Depending on budget, this may be done by a face-to-face meeting, or there may be other ways in which the process can be launched, e.g. via social media. It may be carried out as part of steps 1 and/or 3. The Steering Group should advise on when to do this. Awareness raising has several key objectives:
- to present the proposed plan for the PSP
- to generate support for the process
- to encourage participation in the process
- to initiate discussion, answer questions and address concerns.
Step 3: Identifying evidence uncertainties
The breathlessness PSP will carry out a consultation to gather uncertainties from patients, carers and clinicians. A period of 16 months will be given to complete this exercise (which may be revised by the Steering Group if required).
The breathlessness PSP recognises that the following groups may require additional consideration.
- Digitally excluded or reluctant respondents
- Disadvantaged socio-economic respondents
- Ethnic minorities
- Males
- Children (responses by parent/carer)
- Adolescents (responses by adolescent and/or parent)
- Young adults (18-30yrs)
In order to reach these groups throughout the PSP the following strategies are proposed:
- Attending Asthma + Lung UK Breathe Easy groups in person to facilitate group completion of the survey or completion through a focus group style session
- Placing physical flyers advertising the PSP in breathlessness clinic waiting rooms
- Commissioning the Centre for Ethnic Health Research at the University of Leicester
- Approaching the Royal College of Paediatrics for Child Health
- Approaching relevant influences on social media
The Steering Group will use the following methods to reach the target groups:
- Online survey with organic marketing (i.e. not paid)
- promotion via mailing lists (that of our own and partner organisations) and Asthma + Lung UK social media
- Online surveys with paid for marketing
- via social media channels
- Potentially focus groups (in person & online) for under-represented demographic categories as listed above
Existing sources of evidence uncertainties may also be searched - none planned at present.
Step 4: Refining questions and uncertainties
The consultation process will produce ‘raw’ questions and comments indicating patients’, carers’ and clinicians’ areas of uncertainty. These raw questions will be categorised and refined by Dr. Hannah Springs into summary questions which are clear, addressable by research, and understandable to all. Similar or duplicate questions will be combined where appropriate. Out-of-scope and ‘answered’ submissions will be compiled separately. The Steering Group will have oversight of this process to ensure that the raw data is being interpreted appropriately and that the summary questions are being worded in a way that is understandable to all audiences. The JLA Adviser will observe to ensure accountability and transparency.
This will result in a long list of in-scope summary questions. These are not research questions and to try and word them as such may make them too technical for a non-research audience. They will be framed as researchable questions that capture the themes and topics that people have suggested.
The summary questions will then be checked against evidence to determine whether they have already been answered by research. This will be done by Dr. Hannah Springs . The PSP will complete the JLA Question Verification Form, which clearly describes the process used to verify the uncertainty of the questions, before starting prioritisation. The Question Verification Form includes details of the types and sources of evidence used to check uncertainty. The Question Verification Form should be published on the JLA website as soon as it has been agreed to enable researchers and other stakeholders to understand how the PSP has decided that its questions are unanswered, and any limitations of this.
Questions that are not adequately addressed by previous research will be collated and recorded on a standard JLA template by Dr. Hannah Springs. This will show the checking undertaken to make sure that the uncertainties have not already been answered. The data should be submitted to the JLA for publication on its website on completion of the priority setting exercise, taking into account any changes made at the final workshop, in order to ensure that PSP results are publicly available.
The Steering Group will also consider how it will deal with submitted questions that have been answered, and questions that are out of scope.
Step 5: Prioritisation – interim and final stages
The aim of the final stage of the priority setting process is to prioritise through consensus the identified uncertainties about breathlessness. This will involve input from patients, carers and clinicians. The JLA encourages PSPs to involve as wide a range of people as possible, including those who did and did not contribute to the first consultation. There are usually two stages of prioritisation.
- Interim prioritisation is the stage where the long list of questions is reduced to a shorter list that can be taken to the final priority setting workshop. This is aimed at a wide audience, and is done using similar methods to the first consultation. With the JLA’s guidance, the Steering Group will agree the method and consider how best to reach and engage patients, carers and clinicians in the process. The most highly ranked questions (around 25) will be taken to a final priority setting workshop. Where the interim prioritisation does not produce a clear ranking or cut off point, the Steering Group will decide which questions are taken forwards to the final prioritisation.
- The final priority setting stage is generally a one-day workshop facilitated by the JLA. With guidance from the JLA and input from the Steering Group, up to 30 patients, carers and clinicians will be recruited to participate in a day of discussion and ranking, to determine the top 10 questions for research. All participants will declare their interests. The Steering Group will advise on any adaptations needed to ensure that the process is inclusive and accessible.
Dissemination of results
The Steering Group will identify audiences with which it wants to engage when disseminating the results of the priority setting process, such as researchers, funders and the patient and clinical communities. They will need to determine how best to communicate the results and who will take responsibility for this. Previous PSPs’ outputs have included academic papers, lay reports, infographics, conference presentations and videos for social media.
It should be noted that the priorities are not worded as research questions. The Steering Group should discuss how they will work with researchers and funders to establish how to address the priorities and to work out what the research questions are that will address the issues that people have prioritised. The dissemination of the results of the PSP will be led by Dr. Erika Kennington.
The JLA encourages PSPs to report back about any activities that have come about because of the PSP, including funded research. Please send any details to the JLA.
Agreement of the Steering Group
The breathlessness PSP Steering Group agreed the content and direction of this Protocol on 1st November 2023.