Lung Transplantation (Canada) PSP protocol

Purpose of the PSP and background

The purpose of this protocol is to outline the aims, objectives, methods, and commitments of the Lung Transplant Priority Setting Partnership (PSP) in line with James Lind Alliance (JLA) principles and the basic roles and responsibilities of the partners.

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.

Lung transplantation is a surgical procedure done to replace a diseased or failing lung with a healthy lung from a deceased donor. It is the main therapeutic option considered for end-stage lung diseases, which can not only improve survival but also the quality of life for patients and their caregivers. In the International Society of Heart and Lung Transplantation Registry, the most common lung diseases requiring lung transplantation are interstitial lung disease (including idiopathic pulmonary fibrosis), Chronic Obstructive Pulmonary Disease (COPD)/ emphysema, cystic fibrosis and other rare lung diseases. With life expectancy for people with end-stage lung conditions being 2-4 years, lung transplantation offers a better survival rate and quality of life for such patients. Lung transplant referrals are made when all other treatment options have been explored, the patient survival is less than 2 years without the transplant and poor quality of life/ inability to perform daily needs.

Due to limited availability of donor lungs, some patients who are eligible for transplantation will die on the wait list. Although lung transplantation improves survival and quality of life for most patients, some experience major complications from the surgery itself and others suffer complications related to immunosuppression with anti-rejection drugs, including infections, increased risk of cancer, other organ dysfunction, and rejection of the transplanted lung. Overall, the expected survival after lung transplantation internationally is 85% at 1 year and, of those surviving the first year, 70% will survive to 5 years post-transplant, which is shorter than all other solid-organ transplants (e.g., kidney, liver, heart).

To improve outcomes for patients receiving lung transplants, more research will be needed. There are many potential avenues for future research benefiting lung transplant recipients, but the priorities for future research according to patients, caregivers and clinicians are not defined.

Aims, objectives and scope of the PSP

The aim of the Lung Transplantation PSP is to identify the unanswered questions about lung transplantation 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 overall aims of this PSP are to identify gaps in the literature and prioritise evidence uncertainties.

The Lung Transplantation PSP will identify the relevant uncertainties that reflect questions, or the difficulties faced by patients, their family, caregivers, specialists and clinicians.  This process will highlight the important problems pre- and post-transplant, including but not limited to psychosocial issues, rehabilitation, diagnosis and management complications of lung transplantation.

By identifying the most significant and important health care gaps that need to be addressed, this project will have a lasting impact on the research agenda for years to come. This PSP will also create opportunities for future patient-oriented research involvement for patients, their family and primary caregivers in the design and conduct of clinical research.

The objectives of the PSP are to:

• Work with patients, carers and clinicians to identify uncertainties about lung transplantation
• To engage patients waiting for transplant, patients who have undergone lung transplantation, family members of patients listed for transplantation and who have received transplantation, other caregivers and clinicians in order to identify uncertainties about pre and post operative care, complications that follow, rehabilitation and prognosis of lung transplantation.
• To elicit from those engaged the most important areas of uncertainty.
• To agree by consensus on a prioritised list of those uncertainties, for research
• To measure the impact and value of this initiative to patients & caregivers, clinicians, researchers.
• To ensure a diversity of responses and involvement from underrepresented sample groups and from all provinces and territories in Canada.
• 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 Lung Transplantation PSP is defined as:

• Adult pre-, and post-lung transplantation
• Canadian patients, caregivers, and clinicians at all lung transplantation surgical centres (Vancouver, Edmonton, Toronto, Montreal) as well as satellite centres that follow pre- and post-transplant patients.
• Uncertainties relating to the assessment of patients for lung transplantation eligibility, bridging patients to transplant, potential donor management and donor organ procurement practices, donor organ optimization (i.e. ex-vivo or ex-situ lung perfusion), intraoperative management and surgical approaches, monitoring and diagnosis of post-transplant complications, rehabilitation, treatment of transplant-related complications, critical care management of early and late transplant complications, service organisation and delivery

We will ensure that underserved populations in Canada, specifically Indigenous patients and carers and new Canadians, are purposefully engaged.

The PSP will exclude from its scope questions about:

• Paediatric lung transplantation
• Questions related to donation advocacy and health policy

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 Lung Transplantation PSP will be led and managed by a Steering Group involving the following:

Patient and caregiver representative/s

1. Eileen Leopold- Caregiver representative
2. Rowan Sargeant – Patient representative

Clinical representative/s

1. Margret Michaels, MD- Masters Student, University of Alberta
2. Jason Weatherald, MD- University of Alberta
3. Kieran Halloran, MD- University of Alberta
4. Nikki Marks, Nurse Practictioner (University Health Network, Toronto)
5. Basil Nasir, MD (Transplant Surgeon - Université de Montréal)
6. Celine Bergeron, MD (University of British Columbia)
7. Lea Harper, MD (University of Calgary - satellite/non-surgical centre)

Project coordinators

1. Margret Michaels, MD- Masters Student, University of Alberta
2. Jason Weatherald, MD- Supervisor, University of Alberta

James Lind Alliance Adviser and Chair of the Steering Group

Tamara Rader - JLA Adviser

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 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:

• Lung Transplant patients/ candidates
• Caregivers of people who have had lung transplant
• Family members of people who have had lung transplant
• Patient advocacy groups and professional societies (e.g., Canadian Society for Transplantation, Canadian Donation and Transplantation Research Program, International Society of Heart and Lung Transplantation)
• Clinicians involved in care of patients with lung transplants (e.g., nurses, physicians, surgeons, intensive care unit staff, physiotherapists, pharmacists)
• Health system administrators (Alberta Health Services) and health care professionals (both with experience in lung transplant)

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 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 Lung Transplantation PSP.

The Steering Group would be responsible for drafting the invitation, and an agreement should be reached as to the best organisation to distribute it.

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. This will be done via the Lung Transplantation PSP website, emails to potential partner organisations (e.g., Canadian Society of Transplantation), social media accounts and by engaging patients and clinicians at Lung Transplantation clinics across the country.

The awareness raising will have several key objectives:

• To welcome and introduce potential members of the Lung Transplantation PSP
• To present the proposed plan for the PSP
• To generate support for the process
• To initiate discussion, answer questions and address concerns
• To identify those potential partner organisations which will commit to the PSP and identify individuals who will be those organisations’ representatives and the PSP’s principal contacts
• To establish principles upon which an open, inclusive and transparent mechanism can be based for contributing to, reporting and recording the work and progress of the PSP
• 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 Lung Transplantation PSP will carry out a consultation to gather uncertainties from patients, carers and clinicians. A period of 5 months will be given to complete this exercise, (which may be revised by the Steering Group if required).

The PSP Steering Group will be tasked to promote to its members an online survey asking for evidence uncertainties relating to lung transplantation from patients, caregivers, family members of living and deceased lung transplantation patients, and clinicians.

The Steering Group will use the following methods to reach the target groups

• online surveys
• email invitations to complete the online survey
• direct contact with patients, caregivers and clinicians at lung transplantation clinics across Canada to complete the online survey

The key deliverables will be the development, piloting and distribution of an online initial survey. In the initial survey, an open-ended question will be given and the participants' basic demographic information will be requested in both English and French.

Piloting the survey will occur by distributing them to 5 patients in the Edmonton Lung Transplantation clinic to ensure the purpose and questions are clear and easily understood. The revised survey will be translated to French and reviewed by French speaking clinicians and patients to ensure clarity and correctness.

Each respondent can propose up to 10 questions or uncertainties. We will use a secure REDCap survey platform based at the University of Alberta to capture and store the online survey data.

A link to the online survey will be distributed on the Lung Transplantation PSP website, by email invitations from our partners to their members, on social media (e.g., Twitter) and cards/posters with a QR code that can be given directly to patients and caregivers in clinics. Each patient, caregiver and the families/caregivers of both living and deceased lung transplant patients will be approached in outpatient lung transplantation clinics all across Canada to complete the online survey. The initial survey will remain open for 4 months. Other clinics across Canada may also administer paper surveys and send them by post to the coordinating centre. Most lung transplant patients are followed up intensely in clinics, which will help ensure the survey can be proposed to the majority of the patients in these clinics.

The background information (to be included in the survey form) as well as the wording of the survey items will be designed to be clear and easy to understand by all participants. The following identifiable demographic data will be requested in the survey:

• Age
• Gender
• geographic location (province)
• ethnic group
• The participant is a patient, caregiver or family member, health professional, administrator or professional in a health care system organisation or a member of a partner organisation that represents people who have undergone lung transplantation.

These data will be collected in an effort to determine whether the perspectives of our stakeholder groups of interest have been successfully captured, and to study whether research priorities are influenced by these demographic factors.

Existing sources of information about evidence uncertainties for patients and clinicians will be searched. These can include question-answering services for patients and carers and for clinicians; research recommendations in systematic reviews, literature search and clinical guidelines; protocols for systematic reviews being prepared and registers of ongoing research will be performed to identify additional gaps, uncertainties and research priorities identified in current national and international lung transplantation guidelines.

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 Margret Michaels 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 Margret Michaels under the supervision of Jason Weatherald and Kieran Halloran. A structured search will be developed in consultation with a medical librarian. We will search Medline/PubMed, Embase, and Cochrane database of systematic reviews. Citations will be included if they are systematic reviews, guidelines, randomized controlled trials, or prospective observational studies. Articles that are retrospective studies, editorials, case series, reviews, and case reports will be excluded. Included articles will be mapped to each question to determine if they have been adequately answered. Questions answered based on the results from a systematic review, guidelines with high-quality evidence, and at least one randomized controlled study or two prospective observational studies will be considered sufficient evidence for a question to be deemed certain and will not be considered further for prioritization.

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 Margret Michaels under the supervision of Jason Weatherald and Kieran Halloran. 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 lung transplantation. 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.

1. 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 on 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.
   
   
2. 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 Margret Michaels under the supervision of Jason Weatherald and Kieran Halloran.

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 Lung Transplantation PSP Steering Group agreed the content and direction of this Protocol on October 10, 2023

REFERENCES
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4. Carney KC, Bronzell-Wynder T, Gronek K. Lung Transplant for the Critical Care Nurse. Crit Care Nurs Clin North Am. 2019;31(3):285-302. doi:https://doi.org/10.1016/j.cnc.2019.05.001
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