Polycystic Ovary Syndrome PSP protocol

Purpose of the PSP and background

The purpose of this protocol is to clearly set out the aims, objectives and commitments of the Polycystic Ovary Syndrome (PCOS) 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, family members, 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, family members, 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 is a PSP that is led by a partnership between Cardiff University and Verity, the UK-wide PCOS focussed charity, building on our previous work together on research projects and applications for funding. The funding for the PSP has been provided by the Waterloo Foundation.

PCOS is a   common endocrine (hormone-related) condition affecting women: This includes people assigned female at birth but who no longer identify as female. Through all the documents developed as part of this JLA PSP we will follow the approach to use of language developed in the International PCOS guidelines aimed at harmonising gender inclusivity with biological precision. Therefore we have tried to use accessible and gender-neutral language such as people living with PCOS where possible. For biological clarity we will use terms like “female”. Where the terms woman/women are used they refer to all genders affected by PCOS, however people identify. 

PCOS presents a range of symptoms: Some people with PCOS may experience many of these symptoms, others only a few. The most frequent symptoms include irregular periods, excess facial/body hair, weight gain, acne, scalp hair loss, difficulties getting pregnant, anxiety and depression, reduced quality of life and health complications, including type 2 diabetes and heart disease. Unfortunately, current treatments often prove ineffective, there are high levels of  patient dissatisfaction with care and there is a notable lack of ongoing, high-quality research to improve treatment. As a result, there is an urgent need for research funding organisations to support rigorous investigations in this field. Although information on priorities for research in the international community will soon be published from the recently completed update to the 2023 International PCOS guidelines, no UK equivalent has yet been undertaken. 

Verity will lead the work on stakeholder engagement and administration of the project and the Cardiff University team will lead the academic component of the partnership. Both teams will work together to engage with the Steering Group, develop materials and disseminate the study findings. 

Aims, objectives and scope of the PSP

The aim of the PCOS PSP is first to identify the unanswered questions about PCOS from patient, family member, carer and clinical perspectives and then prioritise the questions that these groups of people agree are the most important for research to address. Carers are an important group in many JLA PSPs:  In relation to PCOS, the term “carer” is used to refer to carers supporting people with non-PCOS conditions that they live with, in addition to PCOS.

The objectives of the PSP are to:

• work with people living with PCOS, their partners/family members/carers and clinicians to identify uncertainties about all aspects of PCOS, including causes, managing symptoms, prevention, diagnosis, how healthcare is provided, access to reliable information, long-term health implications and outcomes (including physical, psychological, social and societal outcomes).
• 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 PCOS PSP is defined as:

• including all aspects of PCOS in the UK context: including causes, managing symptoms, prevention, diagnosis, how healthcare is provided, access to reliable information, long-term health implications and outcomes (including physical, psychological, social and societal outcomes).

The PSP will exclude from its scope questions about:

• health conditions not related to PCOS.
• service provision in non-UK settings. 

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 has been put in place to do this evidence checking. 

The Steering Group

The Steering Group membership includes patients and clinicians, as individuals or representatives from a relevant group.

The PCOS PSP will be led and managed by a Steering Group involving the following:

Patient  representative/s

Ashley Reji - Patient Representative 
Debbie Shaffer - Patient Representative for Fair Treatment for the Women of Wales (FTWW) - Wales
June-Ann Joseph - Patient Representative 
Maureen Busby - PCOS Vitality patient Group - Northern Ireland 
Neelam Heera - Patient Representative for Cysters
Saidah Isse Mohamed - Patient Group - PCOS Society University of Glasgow
Yasmin Nichols -  Patient Representative 

Clinical representative/s

 Adam Balen - Professor of Reproductive Medicine and Surgery, The Leeds Teaching Hospitals NHS Trust
Alison Layton - Consultant Dermatologist, Harrogate and District NHS Foundation Trust
Bassel Wattar - Associate Professor of Reproductive Medicine, Anglia Ruskin University and University College London
Caroline Gillet - Assistant Professor in Patient and Public Involvement and Engagement, University of Birmingham
Grace Dugdale - Nutrition Scientist
Helena Teede - Director of Monash Centre for Health Research Implementation and Endocrinologist,  Monash University, Australia
Jacky Boivin - Professor of Health Psychology, Cardiff University
Jadine Scragg - Researcher,  University of Oxford
Judith Ibison - Professor of Practice - Primary Care, St George's University of London
Punith Kempegowda - Assistant Professor in Endocrinology, Diabetes and General Medicine, University of Birmingham
Steve Franks - Emeritus Professor of Reproductive Endocrinology, Imperial College, London

PSP Project Team

PSP lead: Aled Rees - Professor of Endocrinology, Cardiff University
Project coordinators: Sue Channon - Senior Research Fellow, Cardiff University and
Elinor Coulman - Research Associate, Cardiff University
Project Administrator: Fiona Morgan  - Administrator, Verity
Verity Trustee Representatives: Rachel Morman, Caroline Andrews 
Information Specialist: Jet Hounsome - Cardiff University     
James Lind Alliance Adviser and Chair of the Steering Group: Jo Watson 

 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 PCOS and those with suspected PCOS 
• women’s groups that are not PCOS-specific but potentially provide a voice for people from under-represented communities and people with other PCOS-related conditions
• health and social care professionals with experience of working with PCOS as part of their professional role.

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. The Steering Group will ensure there is representation from across the four nations. Potential partners will be contacted and informed of the establishment and aims of the PCOS PSP.

Step 2: Awareness raising 

PSPs need to raise awareness of their proposed activity among their patient and clinician communities, including the family members of those living with PCOS, in order to secure support and participation.  For the PCOS PSP this will be done via the clinical and patient networks and via social media.  It may be carried out as part of steps 1 and 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 PCOS PSP will carry out a consultation to gather uncertainties from people living with PCOS and clinicians.  A period of four months will be given to complete this exercise (which may be revised by the Steering Group if required). 

Inclusion Criteria for the consultation are:

• Over 16 years of age 
• Living in the UK
• People living with PCOS (with a confirmed or suspected diagnosis)      
• People who support someone living with PCOS (e.g. parent or partner)
• People providing health or social care to people living with PCOS, both within statutory organisations, third sector and private healthcare. 

The PCOS PSP recognises that some groups may require additional consideration e.g those from global majority or socially disadvantaged communities who may be less likely to connect with third sector organisations representing people living with PCOS. 

The PSP team and Steering Group will use the following methods to reach the target groups: 

• Optimise use of Verity’s strong social media presence on X, Instagram, Tiktok and Facebook     
• Develop audiovisual versions of the information and use AI tools for translation where possible to improve accessibility
• Offer on-line or face-to-face discussion with groups to disseminate the information sheets, answer any queries and facilitate access to the survey content.

Existing sources of evidence uncertainties will also be searched, building in particular on the recent work of the international guidelines development groups, and protocols for systematic reviews. 

Ethical Review

The ethics committee in the School of Medicine, Cardiff University have advised that this step of gathering uncertainties from patients and clinicians, and similarly the initial prioritisation survey in step 5 below, aim to derive new, generalisable knowledge which goes beyond this specific project. Therefore, these two tasks in steps 3 and 5, that engage with patients and clinicians beyond the Steering Group members via a survey, meet the threshold definition of research and require approval from the ethics committee. 

Step 4: Refining questions and uncertainties

The consultation process will produce ‘raw’ questions and comments indicating patients’, family members’ and clinicians’ areas of uncertainty. These raw questions will be categorised and refined by the project team 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, that the summary questions are being worded in a way that is understandable to all audiences and reflect the true essence of the uncertainties submitted in the consultation.  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 information specialist, in conjunction with the PSP lead.  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 will 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 information specialist, in conjunction with the PSP lead.  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 – initial and final prioritisation stages 

The aim of the final stage of the priority setting process is to prioritise through consensus the identified uncertainties about PCOS. This will involve input from patients 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. Initial 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 same eligibility criteria.  With the JLA’s guidance, the Steering Group will agree the method and consider how best to reach and engage patients, family members and clinicians in the process. The most highly ranked questions (around 25) will be taken to a final priority setting workshop.  Where the initial 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, family members     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 Verity and Cardiff University. 

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. 

Agreement of the Steering Group

The PCOS PSP Steering Group agreed the content and direction of this Protocol on 14th November 2024.