Purpose of the PSP and background
The purpose of this protocol is to clearly set out the aims, objectives and commitments of the Medically Not Yet Explained Symptoms (MNYES) 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 – www.nihr.ac.uk) coordinates the infrastructure of the JLA to oversee the processes for PSPs, based at the NIHR Evaluation, Trials and Studies Coordinating Centre (NETSCC), University of Southampton.
Medically Not Yet Explained Symptoms (MNYES), represent up to 10-30% presentations in general medicine settings. They can include fatigue, pain, dizziness, irritable bowel syndrome, and functional neurological symptoms (FND). They are often deemed to be stress-related, and comorbid depressive or anxiety disorder occur in approximately 30% of cases. Patients diagnosed with such symptoms, although care may be taken to explain their condition properly, often do not feel that they are taken seriously. They have to deal with high disability as well as with clinicians who may find MNYES hard to manage. They visit many clinics for diagnostic procedures to hear ‘it is all in your head’. Treatments may pay little attention to their patient experiences and have limited effect.
The focus of research on MNYES is often on particular subsets of symptoms, such as chronic pain, chronic fatigue, irritable bowel syndrome, dizziness etc. but lacking a comprehensive view. This has ramifications for patients who visit different clinics for their various symptoms, without sustained improvement.
There are many different terms used for this condition: Persistent Somatic Symptoms, Somatic Symptom and Related Disorders (SSRD), Medically Unexplained Symptoms (MUS), functional symptoms, chronic pain, conversion disorder, Irritable bowel disorder (IBS), dissociative syncope, dissociative neurological symptoms, functional neurological disorder etc. For this PSP, and the common denominator seems to be distress amongst patients, carers and clinicians in the context of conditions that are difficult to explain, the PSP will be termed Medically Not Yet Explained Symptoms.
The European Association of Psychosomatic Medicine has recently published a research agenda in this domain, one of the research priorities being patient preferences for research in this field. There is still relatively little support available for people with Medically Not Yet Explained Symptoms and those who care for them to get involved in setting the research agenda. The purpose of this PSP is to support patients, carers and clinicians in the UK and in the Netherlands in the development of a research agenda with patient knowledge, experience and preferences as the frame of reference. The Anglo-Dutch partnership will additionally enable exploration of international and intercultural differences that might be of interest for future development of services and the co-design of research to develop better treatment and care.
Aims, objectives and scope of the PSP
The aim of the Medically Not Yet Explained Symptoms PSP is to identify the unanswered questions about Medically Not Yet Explained Symptoms (MNYES) 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 aetiology, diagnosis and treatment of MNYES; about organisation of services, and social consequences and costs.
- 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 MNYES PSP is defined as:
- Aetiology, diagnosis and treatment/care of patients with MNYES in the UK and Netherlands, as well as organisation of services, social consequences and long term outcomes including cost implications for patients.
- International exchange and exploration.
- Confirmed topics include (but are not limited to): pain, fatigue, dizziness, functional neurological disorder, bowel symptoms, palpitations and syncope.
- Ages 16 and older are included.
Fatigue is in scope but Chronic Fatigue Syndrome is out of scope as there is another PSP which is addressing this.
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 MNYES PSP will be led and managed by a Steering Group involving the following:
Patient and carer representative/s
Annie Shaw
Scott Spain
Lesley Spain (carer)
Steph Hayle
Rosie Evans
Clinical representative/s
Professor Christina van der Feltz-Cornelis, honorary consultant psychiatry, University of York, TEWV NHS Trust
Dr. James Sampford, consultant psychiatrist, TEWV NHS Trust
Dr. Jennifer Gilligan, Specialty Doctor in Psychiarty, TEWV NHS Trust
Dr. Christopher Price, Clinical Reader in Stroke Medicine, Newcastle University
Dr. Andrew Moriarty, general practitioner
Dr. Arvind Kaul, Consultant rheumatologist, St Georges Hospital and Medical School in London
Dr. Nick Gall, Consultant cardiologist, Kings College Hospital NHS Foundation Trust
Tracey Cunningham, Physiotherapist, TEWV NHS Trust
Dr. Philippa Bolton, Clinical liaison psychiatrist, TEWV NHS Trust
Dr. Miriam Lomas, Principal Clinical psychologist, TEWV NHS Trust
Dr Petros Perros, Consultant Endocrinologist, The Newcastle upon Tyne Hospitals NHS Foundation Trust
Professor Mark Edwards, Professor of Neurology, St Georges University Hospitals NHS Foundation Trust
Ad Vorselaars, occupational physician, Arbo Unie, the Netherlands
Dr. Arjan Videler, psychotherapist, CLGG, Tilburg, the Netherlands
Information specialist
Danielle Varley and Jennifer Sweetman, University of York
Dr. Iman Elfeddali, and Prof. Dr. Wijo Kop, Tilburg University
Project coordinators
Sally Brabyn, University of York, UK
Natalie Smith, University of York, UK
Llinh Nguyen, Clinical Centre for Body, Mind and Health, The Netherlands
James Lind Alliance Adviser and Chair of the Steering Group
Dr Jonathan Gower, UK
Dr. Margot Metz, The Netherlands
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 had MNYES
- carers of people who have had MNYES
- health care professionals - with experience of working with people with MNYES
- Appropriate organisations that bring together and/or advocate for the patient and caregiver groups will be approached
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 MNYES 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 MNYES PSP will carry out a consultation to gather uncertainties from patients, carers and clinicians. A period of 3 months will be given to complete this exercise (which may be revised by the Steering Group if required).
The MNYES PSP recognises that the following groups may require additional consideration.
- Those in institutions
- People without internet access
- Those without sufficient knowledge of, or confidence in, English or Dutch.
The Steering Group will use the following methods to reach the target groups
- Online surveys that can be made bigger if necessary
- Paper surveys for participants who prefer them
- Face-to-face surveying for those who may have difficulty with reading or writing
- Translation
The Steering Group will decide on a strategy and methods for reaching stakeholder groups that may include some or all of these. It may be appropriate for partner organisations to employ their own methods for reaching their members, within the framework developed by the steering group
Existing sources of evidence uncertainties may also be searched. These will include but not be limited to treatment guidelines and systematic reviews, including protocols for systematic reviews, research papers and registers
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 Danielle Varley 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 Danielle Varley. 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 Danielle Varley. 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 medically not yet explained symptoms (MNYES). 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 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 the steering group.
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 MNYES PSP Steering Group agreed the content and direction of this Protocol on 30th April 2020. Updated September 2020. Updated January 2021.