Purpose of the PSP and background
The purpose of this protocol is to clearly set out the aims, objectives, and commitments of the Childhood Physical Disability Priority Setting Partnership (PSP) in line with James Lind Alliance (JLA) principles. The project protocol is a JLA requirement and will be published on the PSP’s page of the JLA website. The Steering Group commits to reviewing the project protocol regularly and sending updates 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.
This PSP focuses on childhood physical disability. There are no existing research priorities related to childhood physical disability. While there is significant data as it relates to typically developing children, there is limited data on children living with disability. Further, much of the research in the area of childhood physical disability has not considered the values and preferences of children living with disability, their caregivers, or their healthcare providers. A PSP was determined necessary to encourage more patient, caregiver, and healthcare provider informed research in this area. Dr. Jordan Sheriko, Medical Director, Paediatric Rehabilitation at IWK Health, Nova Scotia, Canada, and Dr. Sarah Moore, Assistant Professor in the School of Health and Human Performance and Department of Pediatrics at Dalhousie University, Nova Scotia, Canada. This PSP is funded internally by IWK Health. The first Steering Group meeting took place on 17th January 2023. We have proposed that the final priority setting workshop takes place in December 2024.
Aims, objectives, and scope of the PSP
The aim of the Childhood Physical Disability PSP is to identify the unanswered questions about Childhood Physical Disability from patient, caregiver, and healthcare provider perspectives and then prioritise those that patients, caregivers and healthcare providers agree are the most important for research to address.
The objectives of the PSP are to:
- Work with patients, caregivers, and healthcare providers to identify uncertainties about the diagnosis, treatment, daily functioning of children and youth with physical disability.
- Agree by consensus how to rank in importance a list of priority areas and unanswered questions for research.
- Publicise the results of the PSP and process.
- Take the results to research commissioning bodies to be considered for funding.
The scope of the Childhood Physical Disability PSP is defined as:
- Physical disability includes individuals with physical disability resulting in significant physical impairment excluding those primarily affecting vision and auditory impairment. These may include, brain, spinal cord and nervous injuries, muscular dystrophies, and bone and joint that have occurred before the age of 18.
The PSP will exclude from its scope questions about:
- Disorders primarily impacting vision and hearing.
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 patient, caregivers, and healthcare providers , as individuals or representatives from a relevant group.
The Childhood Physical Disability PSP will be led and managed by a Steering Group. Current membership (June 2023) includes:
James Lind Alliance Adviser and Chair of the Steering Group
- Tamara Rader, James Lind Alliance
Leads
- Dr. Jordan Sheriko, Medical Director, Paediatric Rehabilitation, IWK Health
- Dr. Sarah Moore, Assistant Professor, School of Health and Human Performance, Department of Pediatrics, Dalhousie University
Project Coordinators
- Tori MacDonald, MD Candidate, Dalhousie University
- Sofia So, MSc Candidate, Dalhousie University
Information Specialist
- Sofia So, MSc Candidate, Dalhousie University
Patient and Caregiver Representatives
- Van Bernard
- Peyton Given
- Samantha Horne
- Jeremy Macdonald
- Brad Given
- Julie Clegg
- Meredith Tasiopoulos
Healthcare Provider Representatives
- Colin Hood, Provincial Children with Complex Needs Coordinator, Health PEI
- Theresa Rogers, Coordinator, Patient Experience Office, IWK Health
- Krista Sweet, Manager of Rehabilitation Services, IWK Health
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:
- People who have lived experience of a childhood-onset physical disability
- Family, friends, and caregivers of children who live with physical disability
- Healthcare professionals who work with children who live with physical disability
- Other physical disability support organizations, such as Muscular Dystrophy Atlantic, March of Dimes, Easter Seals, The Shriners, Club Inclusion, Camp Brigadoon, amongst others
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 Childhood Physical Disability 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. This will be done via social media, e.g. Facebook groups, Instagram and via posters in clinics. 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 Childhood Physical Disability PSP will carry out a consultation to gather uncertainties from patients, caregivers, and healthcare providers. A period of 18 months will be given to complete this exercise (which may be revised by the Steering Group if required).
The Childhood Physical Disability PSP recognises that the following groups may require additional consideration. We may require extra methods for reaching children with a physical disability as this has been a hard-to-reach population for research.
The Steering Group will use the following methods to reach the target groups:
- Social media (e.g., Facebook, Twitter, Instagram)
- Clinic poster boards, electronic monitors
- Clinic newsletters, listservs
- Supporting disability organizations social media, newsletters, listservs
- Paper-based surveys and electronic surveys
- Face-to-face workshops
Existing sources of evidence uncertainties may also be searched. Literature searches on various databases will be conducted to identify already identified evidence uncertainties, true uncertainties, and the current trends in Paediatric Rehabilitation medicine in Canada and globally.
Step 4: Refining questions and uncertainties
The consultation process will produce ‘raw’ questions and comments indicating patients’, caregivers, and healthcare providers’ areas of uncertainty. The PSP acknowledges that not all respondents will submit responses in question format; these responses will be included in the refining process. These raw questions will be categorised and refined by Tori MacDonald and Sofia So 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 and may then be developed into research questions at the end of the priority setting exercise.
The summary questions will then be checked against evidence to determine whether they have already been answered by research. This will be coordinated by the Project Leads with support of the Steering Group. 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 Tori MacDonald or Sofia So. 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 Childhood Physical Disability. This will involve input from patients, caregivers, and healthcare providers. 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, caregivers, and healthcare providers 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 two * half-day workshops through an online meeting platform (e.g., Zoom) and/or in-person meeting, facilitated by the JLA. With guidance from the JLA and input from the Steering Group, up to 30 patients, caregivers, and healthcare providers 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. Children will also be involved in the final priority setting; a workshop will take place for children, which occur either alongside the workshop for caregivers and professionals or beforehand.
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 patient, caregivers, and healthcare provider communities. The Steering Group will 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 the Project Leads.
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
This project protocol was agreed upon by the Steering Group on March 7, 2023.
Published on the JLA website May 30, 2023
Protocol Amended March 18, 2024