ME CFS PSP Protocol

Contents

Context

The purpose of this protocol is to clearly set out the aims, commitments, and plan for the Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS) Priority Setting Partnership (PSP).  

The James Lind Alliance (JLA) is a non-profit making initiative, established in 2004.  It brings together patients, carers, and clinicians in PSPs.  These PSPs identify and prioritise the ‘unanswered questions’ that they would like to see answered by research, using the JLA approach.

The National Institute for Health Research (NIHR – www.nihr.ac.uk) coordinates the infrastructure of the JLA.  The JLA support team is based at the NIHR Evaluation, Trials and Studies Coordinating Centre (NETSCC), University of Southampton.

Further details about the JLA and PSPs are at http://www.jla.nihr.ac.uk/.  The JLA Guidebook and useful templates and examples are available on the website for viewing and downloading.

The PSP has been formed with the support of Action for ME, Forward ME, the Science4ME Forum and the Patient Advisory Group to the UK CFS/ME Research Collaboration. With funding from the National Institute for Health Research, the Scottish Government Chief Scientist Office and the Medical Research Council

Aims, objectives

The aim of the PSP is to identify, prioritise and raise awareness of the unanswered questions which are the most important for research to address from the perspective of people in this community.

The objectives are to:

  • Gather questions through wide consultation from people with ME/CFS, carers, and the clinicians who look after them.
  • Use the JLA process to prioritise and create a shared consensus of the questions which really matter to this community.
  • Publicise the results of the PSP to raise awareness and to encourage research and funding.

The scope

The scope aims to be inclusive and transparent. ME/CFS is a long-term (chronic), fluctuating, neurological condition that causes symptoms affecting many body systems, more commonly the nervous and immune systems. ME/CFS affects an estimated 250,000 people in the UK, and around 17 million people worldwide.

The World Health Organization has classified ME as a neurological disease since 1969. The underlying mechanisms of the condition are not known however people with ME/CFS experience severe, persistent symptoms associated with post-exertional malaise (the body’s inability to recover after expending even small amounts of energy).

For the purposes of identifying research questions, we will assess existing research using well-accepted diagnostic criteria: the Institute of Medicine (IoM) 2015 or the 2003 Canadian Consensus Criteria (CCC), but not the Oxford or NICE criteria. Studies with other criteria will be considered only where post-exertional malaise (PEM) is considered a mandatory symptom. This is because patients, patients’ organisations, and ME/CFS biomedical researchers all regard it as a defining symptom of the disease.

Target audience includes:

  • people with ME/CFS diagnosed by a clinician;
  • people who have had ME/CFS;
  • all of UK;
  • ages 16 and older;
  • carers of younger children with ME/CFS.

The Steering Group trusts that people participating in the project have a clinical diagnosis of ME/CFS, or they are the supporter or a clinician for a person with ME/CFS.  This does mean that someone can participate even if they do not have a clinically verified diagnosis. However, the number of people who might do so is not expected to impact the process,  and it is preferable to keep any barriers for participation to a minimum.

The questions may be about:

  • causes, prevention, risk factors, diagnosis;
  • living with and disability associated with ME/CFS;
  • symptoms, relapses, treatment or management;
  • issues for carers;
  • services relevant to the condition and access to services.

The scope of the PSP excludes:

  • People without a diagnosis of ME/CFS (or CFS/ME or CFS or ME)
  • The symptom of Chronic Fatigue caused by other conditions is excluded.
  • Studies of ME/CFS using the Oxford Criteria, NICE 2007 criteria, Fukuda Criteria without mandatory PEM.

The scope may have implications for the amount of data that will be collected, and how this can be analysed. 

The Steering Group (SG)

The Steering Group includes balanced numbers of people with ME/CFS, carers and clinicians, as individuals or representatives from a relevant group. The members' details are recorded in Annex A.

The SG directs and oversees the PSP.  The role of the members is described in detail in the Terms of Reference.  The SG is facilitated by an independent, neutral JLA Adviser.

Partners

Partners are identified by SG members. Partners are individuals or organisations or groups who can reach and advocate for people who the SG would like to participate in the PSP. Partners commit to supporting the PSP, promoting the process and encouraging their members to participate, but have no role on the SG. 

Partners represent the following groups:

  • people who have, or have had ME/CFS
  • supporters of people who have, or have had ME/CFS
  • health and social care professionals with experience of ME/CFS.

Some organisations may be judged by the JLA or the SG to have a conflict of interest, with potential to bias the process. This can include for example commercial or political interests.  Such organisations will not be invited to participate.  

The methods the PSP will use

Detailed information about the JLA process in available in the JLA Guidebook, www.jla.nihr.ac.uk.

An overview of the steps the PSP will follow is provided here:

Step 1a: Forming the Steering Group (SG)

The Patient Advisory Group, Forward ME and a representative from the S4ME Forum have supported Action for ME in the recruitment of SG members. This has been done by application and interview, to ensure a balanced membership. Further recruitment, if needed, can be agreed by the SG. 

Step 1b: Contact with partner organisations and awareness raising

Organisations may be invited to partner with the PSP. SG members and the SG co-ordinator Sam Bromiley will communicate with networks and communities and partners and engage their support for various stages of the PSP. Partners may change in the course of the PSP. Sam Bromiley will maintain a master list.

Step 2: Gathering unanswered questions

The JLA team of Caroline Whiting, Beccy Maeso and Amy Street, will develop and run the survey, overseen by the SG.

A period of approximately 3 months will be given to complete this exercise. The SG will advise on target groups, and mechanisms to reach them. The aim is to be inclusive.

Existing unanswered questions from guidelines, systematic reviews and research papers may also be included.

Step 3: Analysis of the submitted questions

The analysis will be supported by Kristina Staley, the PSP information specialist. The submitted questions will be analysed and refined into summary questions. The summary questions reflect the intention and language of the original submitted questions, need to be understandable by all, and addressable by research. Out-of-scope and ‘answered’ questions are compiled separately. 

The SG have oversight of this process.

Step 4: Evidence Checking

Step 3 results in a long list of in-scope summary questions, in plain English. These are checked to ensure they are truly unanswered by existing research and in consideration of the exclusion criteria.  

The SG will consider what, if anything, can be done with any questions that have in fact been answered by research, and any questions that are out of scope but might be of interest for another project.

Step 5: Interim prioritisation 

Interim prioritisation of the long list of summary questions will involve input from a wide range of people with ME/CFS, supporters and clinicians, and will be done using similar methods to the first consultation survey. The most highly ranked questions become the basis for the short list of around 20-25 questions. The SG will review this list, and ensure there is a balance of questions prioritised by all stakeholder groups.

Step 6: Final consensus workshop

The short list of 20-25 questions from Step 5 will be taken to the final priority setting consensus workshop.  The SG will consider the best format for conducting this event to ensure that the process is inclusive and accessible, subject to the needs and preferences of the people participating in the event. 

Guided by the SG and the JLA Facilitator, a balanced number (max 30) of people with ME/CFS, carers and clinicians will be recruited to participate in discussions and prioritisation, to determine the top 10 questions for research.  

All participants will be asked to make a Declaration of Interests, to support transparency.

Step 7. Dissemination of results

The SG will identify audiences with which it wants to engage when disseminating the results of the priority setting process, such as researchers, funders and the stakeholder communities.  They will need to determine how best to communicate the results and how this will be taken forward and by whom.

Previous PSPs’ outputs have included academic papers, lay reports, infographics, conference presentations, videos and podcasts for social media.

It should be noted that the priorities are not worded as research questions.  The SG will need to consider how best to support and work with researchers and funders to develop meaningful research questions which will address the priorities.

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 jla@soton.ac.uk

In the interests of transparency and to help contribute to the evidence base, the SG commits to completing the JLA Question Verification Form, the full Datasheet form, and the Engagement Summary form, and submitting these to the JLA, at the end of the PSP.  This will enable researchers and other stakeholders to understand how the PSP has decided that its questions are unanswered, and any limitations of this.

The verified unanswered questions will be recorded on a standard JLA template by Kristina Staley. This will show the verification undertaken for each question to make sure that the question has not already been answered by research, as well as the evidence for those that have been answered.

The full Data template will be completed by Kristina Staley. This will include the derivation of all of the summary questions from the submitted raw questions, including data about which stakeholder groups submitted the questions. It also shows which questions were deemed out of scope, or already answered.  This is a full audit trail, showing how each submitted questions has been reviewed and used.

The Engagement summary records the level of engagement with participants at each stage of the process. This will be completed by Sam Bromiley, supported by Kristina Staley and the JLA team.

Commitment and agreement of the Steering Group

The ME/CFS PSP Steering Group agree the content and direction of this Protocol on Tuesday 8 December 2020.  

Annex A – Members of the Steering Group  

People with ME/CFS:

  • Annette Barclay
  • Sian Leary
  • Monica Bolton
  • Ben Marsh

Carers:

  • Mike Emmans-Dean
  • Natalie Hilliard
  • Ann West
  • Rachel Elliott

Clinicians:

  • Prof Vinod Patel, Professor of Clinical Science at Warwick University
  • Sarah Tyson
  • Joan Crawford, Chartered Counselling Psychologist
  • Tina Betts

The Science 4 ME Forum:

  • Debbie Smith

Forward-ME:

  • Russell Flemming

Patient Advisory Group of the UK CFS and ME Research Collaborative:

  • Representatives from the Patient Advisory Group (PAG) will attend the PSP on a rotating basis.

 

Steering Group support:

  • Sonya Chowdhury, PSP lead, CEO Action for ME
  • Sam Bromiley, PSP coordinator
  • Toto Gronlund, JLA Facillitator
  • Kristina Staley, Information Specialist
  • Beccy Maeso, Caroline Whiting, Amy Street, JLA Secretariat and lead for the surveys