Purpose of the PSP and background
The purpose of this protocol is to clearly set out the aims, objectives and commitments of the UK Epilepsy 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.
Context and background
Epilepsy costs the NHS £1.5 billion a year, and there are over 100,000 emergency admissions due to epilepsy each year. Research into epilepsy receives only 7% of neurological research funding and just 0.3% of the £4.8 billion total spent on health-related research in the UK. At a time when the need to make savings is paramount, there is a compelling case to invest more in research into epilepsy - the return on investment could lead to substantial cost-savings to the NHS and improvements in the treatment and care of people with epilepsy and their families and carers.
Aims, objectives and scope of the PSP
The aim of the UK Epilepsy PSP is to identify the unanswered questions about epilepsy 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 people affected by epilepsy and clinicians to identify uncertainties about causes, diagnosis, treatments and clinical management of epilepsy
- 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.
Definition of epilepsy:
According to the International League Against Epilepsy (ILAE), epilepsy is a disease of the brain defined by any of the following conditions:
- At least two unprovoked (or reflex) seizures occurring >24h apart
- One unprovoked (or reflex) seizure and a probability of further seizures similar to the general recurrence risk (at least 60%) after two unprovoked seizures, occurring over the next 10 years.
- Diagnosis of an epilepsy syndrome
Further information can be found here:
The UK Epilepsy PSP is for people with epilepsy, and therefore does not include those with functional seizures.
The scope of the UK Epilepsy PSP is defined as:
The health research priorities of people with epilepsy to include:
- Causes: to include prevention (including the impact of epilepsy on antenatal and maternal health), co-morbidities
- Diagnosis: to include access to health services
- Treatments: to include anti-epileptic drugs, surgery, treatment side effects, co-morbidities (including those as a consequence of treatment), and epilepsy that does not respond to treatment
- Clinical management of epilepsy: to include risk of epilepsy related deaths (including Sudden Unexpected Death in Epilepsy – SUDEP), service access, neurodevelopment, co-morbidities, social and psychological factors, and emerging areas such as epilepsy in older people
- Dissemination of research breakthroughs to influence epilepsy care, practice and policy: to include medical education and pathways to improved care
The PSP will exclude from its scope questions about:
- Social research
- Health & Social funding
- Non-UK based research
- Functional seizures
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 people affected by epilepsy and clinicians, as individuals or representatives from a relevant group.
The UK Epilepsy PSP will be led and managed by a Steering Group involving the following:
|Ms Alice Yandle||Personal experience of epilepsy|
|Ms Andree Mayne||Epilepsy Society (Patient organisation)|
|Ms Angie Pullen||Epilepsy Action – Director of Epilepsy Services (Patient organisation)|
|Ms Anna Norton||ERUK Research & Involvement Officer (JLA Coordinator)|
|Dr Anne Coxon||Health Psychologist, personal experience of epilepsy|
|Dr Caoimhe Twohig Bennett||ERUK Head of Research (Patient organisation)|
|Professor Hannah Cock||Professor of Epilepsy & Medical Education, Neurologist|
|Dr James Mitchell||Association of British Neurologists Fellow and Neurology Registrar|
|Dr Jon Dickson||General Practitioner with specialist interest|
|Ms Juliet Solomon||Executive Director of the ILAE|
|Ms Katherine Cowan||Senior Adviser, James Lind Alliance (Chair)|
|Ms Maxine Smeaton||ERUK Chief Executive (PSP Sponsor)|
|Dr Michael Kinney||Consultant Neurologist/Epileptologist (Northern Ireland)|
|Mr Phil Tittensor||Chair of ESNA, Epilepsy Clinical Nurse Specialist|
|Dr Rhys Thomas||Neurologist and Neuroscientist (PSP Lead)|
|Professor Rohit Shankar||Professor of Neuropsychiatry, Researcher|
|Ms Rosemarie Pardington||Young Epilepsy, Director of Integrated Care (Patient organisation)|
|Ms Samantha Ashby||SUDEP Action, Deputy Chief Executive (Patient organisation)|
|Professor Sameer Zuberi||Paediatric Neurologist & Researcher|
|Dr Sukhvir Wright||Neurologist and Neuroscientists (Paediatrics)|
|Mr Tom McLaughlan||Personal experience of epilepsy|
|Professor Tony Marson||Professor of Neurology|
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.
Additional 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 epilepsy
- parents, family members and friends of people who have/had epilepsy
- carers of people who have/had epilepsy
- health and social care professionals - with experience of epilepsy.
Database of potential partners
Groups we may struggle to connect with
- people who don’t have English as first language
- people with learning disabilities or limited mental capacity
- BAME community & minority groups
- Health inequalities, social deprivation
- Digitally excluded
- Older adults including people with dementia
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 www.jla.nihr.ac.uk 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 UK Epilepsy 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 UK Epilepsy PSP will carry out a consultation to gather uncertainties from patients, carers and clinicians. A period of 1 year – 18 months will be given to complete this exercise (which may be revised by the Steering Group if required).
The UK Epilepsy PSP recognises that the following groups may require additional consideration.
- There are around 40 different types of epilepsy, with a wide variation in the impact on individuals. A challenge will be ensuring adequate representation from all groups.
- We will also want to consider lower socioeconomic groups as these groups are often linked with higher prevalence of epilepsy
The Steering Group will use the following methods to reach the target
Method 1: Online surveys
Method 2: Publicise surveys through patient organisation networks, neurologists, ESNA, ILAE
Existing sources of evidence uncertainties may also be searched.
- Canadian Epilepsy PSP
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 the PSP Coordinator 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 the PSP Coordinator. 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 the PSP Coordinator. 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 epilepsy. 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 coordinated by Epilepsy Research UK.
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 email@example.com.
Agreement of the Steering Group
The UK Epilepsy PSP Steering Group agreed the content and direction of this Protocol on 12/10/21.