Scoliosis PSP Protocol

Purpose of the PSP and background

The purpose of this protocol is to set out the aims, objectives and commitments of the Scoliosis Priority Setting Partnership (PSP) and the basic roles and responsibilities of the partners therein. It is recommended that the Protocol is reviewed by the Steering Group and updated on at least a quarterly basis.

The James Lind Alliance (JLA) is a non-profit making initiative, established in 2004. It brings patients, carers and clinicians together in Priority Setting Partnerships (PSPs). These partnerships identify and prioritise uncertainties, or ‘unanswered questions’, about the effects of treatments that they agree are the most important. The aim of this is to help ensure that those who fund health research are aware of what really matters to both patients and clinicians. The JLA is coordinated and overseen by the National Institute for Health and Care Research (NIHR) Evaluation, Trials and Studies Coordinating Centre (NETSCC), based in Southampton.

Scoliosis (curvature of the spine) is most common among children and adolescents but also occurs in adults of all ages, with recent research suggesting that 5.9% of females have a curve by the time they reach skeletal maturity, and also linking curvature to an increase in frequency, and severity, of back pain, with more time off school than among youngsters with straight spines. This has an impact on their educational achievement and future employment prospects. Scoliosis is associated with a wide range of other conditions, but in the majority of cases (85%) it is ‘idiopathic’ (i.e. with no known cause). Progression of the condition in children is associated with growth, regardless of cause, whilst in adults progression is associated with larger curves at skeletal maturity, neuromuscular conditions (notably cerebral palsy and various muscular dystrophies) and in those with ‘syndromes’, such as Marfan’s and Neurofibromatosis Type 1. 5% of people with curves will find that they present in the first 2 years of life - some curves can progress rapidly and cause life threatening respiratory and cardiac problems, whilst others will resolve without intervention.

Current diagnostic tests are intrusive and do not always capture the condition, whilst current treatment options can cause body image problems and still leave 40% of people with curves that do not respond and may require surgery. Approximately 2000 scoliosis operations are done each year in England. Surgery has complications, including infection and implant failure leading to revision surgery. The most serious complication is spinal cord damage and paralysis.

There is very little active research on scoliosis in the UK – this Priority Setting Partnership is essential in order to understand what areas for future research are most important to people with scoliosis and the health professionals who work with them.

Aims and objectives of the Scoliosis PSP

The aim of the Scoliosis PSP is to identify the unanswered questions about diagnosis and management of Scoliosis in people of all ages from patient and clinical perspectives, and then prioritise those that patients and clinicians agree are the most important.

For the purposes of this PSP: scoliosis includes all spinal imbalances; diagnosis includes screening and risk of progression; and management includes treatment, care and psychological effects. No age group will be excluded.

Should any cause-related questions be received, these will be extracted from the dataset and passed on to the British Scoliosis Research Foundation and Scoliosis Association UK, to be addressed separately from the PSP

The objectives of the Scoliosis PSP are to:

• work with patients, carers and clinicians to identify uncertainties about the effects of Scoliosis treatments
• to agree by consensus a prioritised list of those uncertainties, for research
• to publicise the results of the PSP and process
• to consider developing one or more of the resulting priorities into a research proposal
• to take the results to research commissioning bodies to be considered for funding.

The Steering Group

The Scoliosis PSP will be led and managed by the following:

Patient and carer/family representative/s:

• Stephanie Clark, Scoliosis Association UK
• Claire Curley, Scoliosis Association UK
• Peter Virley, British Scoliosis Research Foundation
• Fraser Old, Adult Carer representative
• Katie Henderson, Ataxia UK
• Pierre Bernard, Young person Carer representative

Clinical representative/s:

• Andrew Clarke, Consultant Orthopaedic Spine Surgeon, Royal Devon and Exeter
• Jeremy Fairbank, Consultant Spine Orthopaedic Surgeon, Oxford
• Anne Richard, Paediatric Physiotherapist, Oxford University Hospitals NHS Foundation Trust
• Dominique Rothenfluh, Consultant Spinal Surgeon, Oxford University Hospitals NHS Foundation Trust
• Chris Morris, Child Health Group, University of Exeter Medical School

The Partnership and the priority setting process will be supported and guided by:

• Katherine Cowan, The James Lind Alliance (JLA)
• Sandra Regan, The NIHR Oxford Biomedical Research Centre and Musculoskeletal Biomedical Research Unit
• Data Manager/ Information Specialist to be appointed

The Steering Group includes representation of patient/carer groups and clinicians.

The Steering Group will agree the resources, including time and expertise that they will be able to contribute to each stage of the process. The JLA will advise on this.

The wider Partners

Organisations and individuals will be invited to be involved with the PSP as partners. Partners are groups or individuals who will commit to supporting the PSP by disseminating the PSP survey and helping the PSP to gather questions and uncertainties of practical clinical importance relating to the treatment and management of scoliosis. Partners represent the following groups:

• people who have Scoliosis
• carers of people who have Scoliosis
• medical doctors, nurses and professionals allied to medicine with clinical experience of Scoliosis

It is important that all organisations which can reach and advocate for these groups should be invited to become involved in the PSP. The JLA Adviser will take responsibility for ensuring the various stakeholder groups are able to contribute equally to the process.

Exclusion criteria

Some organisations may be judged by the JLA or the Steering Group to have conflicts of interest. These may be perceived to adversely affect those organisations’ views, causing unacceptable bias. 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. For this PSP this includes pharmaceutical organisations, and “pure” researchers with no clinical practice.

The methods the PSP will use

This section describes a schedule of proposed stages through which the PSP aims to fulfil its objectives. The process is iterative and dependent on the active participation and contribution of different groups. The methods adopted in any stage will be agreed through consultation between the Steering Group members, guided by the PSP’s aims and objectives. More details can be found at www.JLAguidebook.org and examples of the work of other JLA PSPs can be seen in the ‘The PSPs’ section of the JLA website at www.jla.nihr.ac.uk

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 Scoliosis PSP and may be invited to attend and participate in an initial stakeholder meeting if this is being arranged.

Step 2: Awareness raising

Awareness raising will be via a dedicated website hosted by the Oxford University Hospitals Foundation Trust under their agreement with the NIHR Oxford Biomedical Research Centre James Lind Alliance “hub”. This will be linked to partner websites and can provide links to partner sites in return

Step 3: Identifying treatment uncertainties

Each partner will identify a method for soliciting from its members questions and uncertainties of practical clinical importance relating to the diagnosis and management of Scoliosis. A period of up to 3 months will be given to complete this exercise.

The methods may be designed according to the nature and membership of each organisation, but must be as transparent, inclusive and representative as practicable. Methods may include membership meetings, email consultation, postal or web-based questionnaires, internet message boards and focus group work.

For this PSP, methods may include: reaching young people via social media, including circa 12,000 members of Scoliosis Association UK via their Facebook page, Instagram and twitter; flyers and electronic screens in clinics; focus groups; and online survey. The possibility of a separate questionnaire for young people was discussed.

Existing sources of information about diagnosis and management uncertainties for patients and clinicians will be searched. These can include question-answering services for patients and carers and for clinicians; research recommendations in systematic reviews and clinical guidelines; protocols for systematic reviews being prepared and registers of ongoing research.

The starting point for identifying sources of uncertainties and research recommendations is NHS Evidence: www.evidence.nhs.uk.

Step 4: Refining questions and uncertainties

The Steering Group will need to have agreed exactly who will be responsible for this stage – the JLA can advise on the amount of time likely to be required for its execution. The JLA will participate in this process as an observer, to ensure accountability and transparency.

The consultation process will produce “raw” unanswered questions about diagnosis and the effects of treatments. These raw questions will be assembled and categorised and refined by [insert name] into “collated indicative questions” which are clear, addressable by research and understandable to all. Similar or duplicate questions will be combined where appropriate.

Systematic reviews and guidelines will be identified and checked by [insert name]to see to what extent these refined questions have, or have not, been answered by previous research. Sometimes, uncertainties are expressed that can in fact be resolved with reference to existing research evidence - ie they are "unrecognised knowns" and not uncertainties. If a question about treatment effects can be answered with existing information but this is not known, it suggests that information is not being communicated effectively to those who need it. Accordingly, the JLA recommends strongly that PSPs keep a record of these 'answerable questions' and deal with them separately from the 'true uncertainties' considered during the research priority setting process.

Uncertainties which are not adequately addressed by previous research will be collated and recorded on a template supplied by the JLA) by [insert name]. This will demonstrate the checking undertaken to make sure that the uncertainties have not already been answered. This is the responsibility of the Steering Group, which will need to have agreed personnel and resources to carry this accountability. 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.

Step 5: Prioritisation – interim and final stages

The aim of the priority setting process is to prioritise through consensus the identified uncertainties relating to the treatment or management of Scoliosis. This will be carried out by members of the Steering Group and the wider partnership that represents patients and clinicians.

• The interim stage, to proceed from a long list of uncertainties to a shorter list to be discussed at the final priority setting workshop (e.g. up to 30), may be carried out over email or online, whereby organisations consult their membership and choose and rank their top 10 most important uncertainties. There are examples of how other PSPs have achieved this at www.jla.nihr.ac.uk in the Key Documents of the Anaesthesia and Perioperative Care PSP section and the Childhood Disability PSP section.
• The final stage, to reach, for example, 10 prioritised uncertainties, is likely to be conducted in a face-to-face meeting, using group discussions and plenary sessions.
• The methods used for this prioritisation process will be determined by consultation with the partner organisations and with the advice of the JLA Adviser. Methods which have been identified as potentially useful in this process include: adapted Delphi techniques; expert panels or nominal group techniques; consensus development conference; electronic nominal group and online voting; interactive research agenda setting and focus groups.

The JLA will facilitate this process and ensure transparency, accountability and fairness. Participants will be expected to declare their interests in advance of this meeting.

Dissemination of findings and research

Findings and research

It is anticipated that the findings of the Scoliosis PSP will be reported to funding and research agenda setting organisations such as the NIHR and the major research funding charities including via the Association of Medical Research Charities (AMRC). Steering Group members and partners are expected to develop the prioritised uncertainties into research questions, and to work to establish the research needs of those unanswered questions to use when approaching potential funders, or when allocating funding for research themselves, if applicable.

Publicity

As well as alerting funders, partners and Steering Group members are encouraged to publish the findings of the Scoliosis PSP using both internal and external communication mechanisms such as via social media and Healthwatch. The Steering Group may capture and publicise the results through descriptive reports of the process itself in Plain English. This exercise will be distinct from the production of an academic paper, which the partners are also encouraged to do. However, production of an academic paper should not take precedence over publicising of the final results.

Agreement of the Steering Group

Signed by the Steering Group

The undersigned agree to follow the Scoliosis Priority Setting Protocol.

Pierre Bernard, Young person Carer representative
Stephanie Clark, Scoliosis Association UK
Claire Curley, Scoliosis Association UK
Peter Virley, British Scoliosis Research Foundation
Fraser Old, Adult Carer representative
Katie Henderson, Patient Representative, Ataxia UK
Andrew Clarke, Consultant Orthopaedic Surgeon, Royal Devon and Exeter
Jeremy Fairbank, Consultant Orthopaedic Surgeon, Oxford
Chris Morris, Child Health Group, University of Exeter Medical School
Anne Richard, Paediatric Physiotherapist, Oxford University Hospitals NHS Foundation Trust
Dominique Rothenfluh, Consultant Spinal Surgeon, Oxford University Hospitals NHS Foundation Trust
Katherine Cowan, The James Lind Alliance