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Research priority setting in plastic and reconstructive surgery: A systematic review

  • Alice Lee
    Correspondence
    Corresponding author.
    Affiliations
    Department of Surgery and Cancer, Imperial College London, London, SW7 2AZ, United Kingdom

    Department of Plastic Surgery, Stoke Mandeville Hospital, Aylesbury, HP21 8AL, United Kingdom
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  • George Higginbotham
    Affiliations
    School of Physiology, Pharmacology and Neuroscience, Biomedical Sciences Building, University Walk, University of Bristol, Bristol, BS8 1TD, United Kingdom
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  • Philippa Davies
    Affiliations
    Population Heath Sciences, Bristol Medical School and Bristol Biomedical Research Centre and Centre for Surgical Research, University of Bristol, BS8 2BN, United Kingdom
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  • Author Footnotes
    # This article is dedicated to Professor Amber Young.
    Amber Young
    Footnotes
    # This article is dedicated to Professor Amber Young.
    Affiliations
    Population Heath Sciences, Bristol Medical School and Bristol Biomedical Research Centre and Centre for Surgical Research, University of Bristol, BS8 2BN, United Kingdom
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  • Author Footnotes
    # This article is dedicated to Professor Amber Young.
Open AccessPublished:October 17, 2022DOI:https://doi.org/10.1016/j.bjps.2022.10.035

      Summary

      Background

      The health research agenda has historically been led by researchers; however, their priorities may not necessarily align with those of patients, caregivers and clinicians. Research priority setting initiatives identify and prioritise topics which lack evidence. This is particularly important in plastic surgery, a speciality lacking high-quality evidence to definitively answer many common clinical questions. Research priorities direct research activity and funding, so their selection process must be representative and transparent. This review appraised all priority setting initiatives in plastic surgery using the reporting guideline for priority setting of health research (REPRISE).

      Methods

      OVID Medline, EMBASE, CINAHL and the James Lind Alliance (JLA) repository were searched (inception - 11/06/21) using search terms for ‘research priority setting’ and ‘plastic and reconstructive surgery’. Dual-author screening and data extraction were conducted, according to PRISMA.

      Results

      Of 3899 de-duplicated citations, 17 were included. Most studies were conducted in national (14/17), high-income (16/17) settings. More priority setting initiatives focussed on burns (6/17) and hand surgery (4/17) than other subspecialties. The JLA (5/17) and qualitative (5/17) approaches were most used for prioritisation, followed by Delphi techniques (3/17), other surveys (3/17) and mixed methods (1/17). A minority included patient (8/17) or multi-disciplinary (8/17) stakeholders. Few reported strategies for implementing research priorities (6/17) or measuring their impact (2/17).

      Conclusions

      Stakeholders from lower-income countries are underrepresented in priority setting initiatives for plastic surgery, despite the global burden of disease. Future studies should recruit more patient and multidisciplinary stakeholders, to achieve meaningful consensus. Clear implementation strategies are needed to maximise impact.

      Keywords

      Introduction

      The health research agenda has historically been led by researchers; however, their research priorities may not necessarily align with those of patients, caregivers and clinicians delivering patient care.
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      Compared with other surgical specialties, plastic and reconstructive surgery particularly lacks high-quality evidence to definitively answer many common clinical questions.
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      This has generated many clinical uncertainties for research prioritisation and to date, various priority setting initiatives have been delivered in areas such as hand and upper limb surgery,
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      congenital defects,

      The ‘top 12’ priorities for research in Cleft Lip and Palate. James Lind Alliance Cleft Lip & Palate Priority Setting Partnership. Available from: https://www.jla.nihr.ac.uk/priority-setting-partnerships/diabetes-type-1/cleft-lip-and-palate/top-10-priorities/.

      skin surgery,
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      The hidradenitis suppurativa priority setting partnership.
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      breast surgery.
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      and aesthetic surgery.
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      ,

      NIHR. NIHR James Lind Alliance Priority Setting Partnerships rolling call. Available from: https://www.nihr.ac.uk/documents/nihr-james-lind-alliance-priority-setting-partnerships-rolling-call/28569.

      so it is important that their selection process is transparent, representative and adequately reported.
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      Tong A., Synnot A., Crowe S., et al. Reporting guideline for priority setting of health research (REPRISE). 2019;3:1–11. doi:10.21203/rs.2.14215/v1.

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      and lack of patient involvement.
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      The aim of this systematic review is to describe the scope, methodology and reporting quality of priority setting initiatives in plastic and reconstructive surgery, using the reporting guideline for priority setting of health research (REPRISE) checklist.

      Tong A., Synnot A., Crowe S., et al. Reporting guideline for priority setting of health research (REPRISE). 2019;3:1–11. doi:10.21203/rs.2.14215/v1.

      The results will inform future researchers conducting research prioritisation in plastic surgery by summarising previous work, identifying areas that are still in need of prioritisation and highlighting both common deficiencies and examples of best practice in the methodology of previous initiatives.

      Methods

      This systematic review adheres to a pre-specified protocol (see supplemental digital content 1) and is reported in line with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses statement.
      • Moher D.
      • Liberati A.
      • Tetzlaff J.
      • Altman D.G.
      Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement.
      It was ineligible for PROSPERO registration as there are no direct health outcomes.

      Identification of studies

      Study eligibility

      Inclusion and exclusion criteria are shown in Table 1. Included studies were full-length peer-reviewed research articles describing research priority setting initiatives in plastic and reconstructive surgery, from database inception to 11th June 2021. Protocol studies were excluded because they lacked information on outcomes. If a research publication describing the priority setting initiative was not available, formal reports from the James Lind Alliance (JLA) website (or equivalent organisation) were used. Multiple publications describing the same research priority setting initiative were merged as one study.
      Table 1Inclusion and exclusion criteria.
      Inclusion criteriaExclusion criteria
      • Studies which elicited stakeholder priorities for research in plastic and reconstructive surgery
      • Full length, peer-reviewed, English language articles
      • Studies using the following methods for identifying research priorities: consensus methods (such as Delphi technique), workshops, consensus conferences, surveys and qualitative approaches (interviews and focus groups)
      • Published from database inception to 11th June 2021
      • Studies irrelevant to plastic and reconstructive surgery
      • Studies assessing priorities for practice and policy (quality indicators), not research
      • Non-original research (literature reviews, policy documents, clinical guidelines, editorials and commentaries) or basic science research
      • Protocol studies
      • Non-English language articles

      Types of participants

      All eligible research priority setting initiatives were included, regardless of type of participant. Stakeholders were defined as patients, carers, healthcare providers, researchers, policy makers and industry representatives.

      Tong A., Synnot A., Crowe S., et al. Reporting guideline for priority setting of health research (REPRISE). 2019;3:1–11. doi:10.21203/rs.2.14215/v1.

      Types of interventions

      Included studies elicited research priorities from stakeholders in areas related to plastic and reconstructive surgery, as defined by the Royal College of Surgeons.

      Plastic and Reconstructive. News & events. Media background briefings and statistics. Royal College of Surgeons. Available from: https://www.rcseng.ac.uk/news-and-events/media-centre/media-background-briefings-and-statistics/plastic-and-reconstructive/.

      Types of outcome

      For all included studies, the primary outcome was a final prioritised list of research topics or questions related to plastic and reconstructive surgery.

      Search strategy

      The following electronic databases were searched from inception to 11th June 2021: OVID Medline, OVID EMBASE and CINAHL. The JLA website was also searched for reports of relevant priority setting partnerships. A search string was developed to identify relevant papers, which included key search terms and medical subject headings for ‘research priority setting’ and ‘plastic and reconstructive surgery’
      • Odgers H.L.
      • Tong A.
      • Lopez-Vargas P.
      • et al.
      Research priority setting in childhood chronic disease: a systematic review.
      ,
      • Graham L.
      • Illingworth B.J.G.
      • Showell M.
      • et al.
      Research priority setting in women's health: a systematic review.
      The database search strings can be found in the supplemental digital content.

      Study selection process

      Search results were combined and de-duplicated using Covidence (Veritas Health Innovation Ltd., Melbourne, Australia). Articles were screened independently by two authors (AL and GH) in two stages (by title and abstract, and full text), according to pre-specified inclusion criteria (Figure 1). A third author (AY) was consulted if discrepancies in article screening could not be resolved.

      Quality assessment

      There is currently no risk of bias assessment tool for studies of research priority setting, and tools designed for trials and observational studies are not applicable. The REPRISE reporting guideline was used for data extraction and to assesses reporting quality.

      Tong A., Synnot A., Crowe S., et al. Reporting guideline for priority setting of health research (REPRISE). 2019;3:1–11. doi:10.21203/rs.2.14215/v1.

      This checklist included items covering the context and scope of the priority setting initiative, governance and team members, stakeholder recruitment and characteristics, identification and prioritisation of research topics, dissemination of results, implementation and evaluation, and disclosures of funding and conflicts of interest.

      Data extraction

      Data were extracted using a piloted data extraction form (Microsoft Excel) developed for the purposes of this review. Data were extracted independently by two authors (AL and GH). A third author (AY) was consulted if discrepancies in data extraction could not be resolved. Studies were categorised into eight subspecialty areas, adapted from the Royal College of Surgeons classification,

      Plastic and Reconstructive. News & events. Media background briefings and statistics. Royal College of Surgeons. Available from: https://www.rcseng.ac.uk/news-and-events/media-centre/media-background-briefings-and-statistics/plastic-and-reconstructive/.

      including congenital conditions, breast surgery, skin surgery, trauma, burns, hand and upper limb surgery, aesthetic surgery and other cancer-related reconstruction (e.g., head and neck cancer, sarcoma or perineal malignancy). Studies were defined as international if their stakeholder groups were multinational. Income status of the research setting (as stated in the text or affiliated with the corresponding author) was described according to the World Bank. The World Bank categorises income status into the following: high, upper-middle, lower-middle and low income countries, based on Gross National Income per capita. Studies were aggregated by the year of publication, pre- or post-2004, when the JLA was established.
      • Partridge N.
      • Scadding J.
      The James Lind Alliance: patients and clinicians should jointly identify their priorities for clinical trials.
      Stakeholders were regarded multidisciplinary if more than one health profession (e.g., surgeon, nursing, therapy, etc.) was included.

      Data synthesis

      Information relating to priority setting context and scope, stakeholder characteristics and study methodology was tabulated (Tables 2-3). Reporting quality (compliance with the REPRISE checklist) is summarised in Table 4 and Figure 2. Table S1 shows a detailed breakdown of reporting compliance with REPRISE per study. A list of included studies can be found in supplemental digital content 2. Based on compliance with the REPRISE checklist and author consensus, a summary of recommendations for future priority setting exercises in plastic surgery is shown in Table 5.
      Table 2Characteristics of included studies.
      N studies
      Geographical scope
      National14
      International3
      Country
      Does not total 17 due to international studies.
      United Kingdom8
      United States7
      Canada3
      Netherlands1
      Norway1
      Australia1
      India1
      World Bank income status
      High16
      Lower middle1
      Royal College of Surgeons Plastic Surgery Subspecialty area
      Burns
      In the original RCS classification, burns are included in the ‘trauma’ subspecialty group.
      6
      Hand and upper limb surgery4
      Skin2
      Congenital1
      General Plastic and Reconstructive Surgery2
      Breast surgery1
      Aesthetic surgery1
      Year of publication
      The JLA was established in 2004.
      Pre-20044
      Post-200413
      Type of prioritised research questions
      Inclusion of question type per study, not per question; does not total 17 as some studies included multiple question types.
      Treatment (intervention)13
      Health services8
      Aetiology6
      Psychosocial6
      Education5
      Economic evaluation4
      Prevention4
      Quality of life4
      Prognosis3
      Diagnosis2
      Stakeholders
      Unclear in n = 1 study.
      Surgeons/doctors9
      Nurses7
      Allied health professionals10
      Patients and care givers8
      Researchers1
      Industry representatives1
      Other6
      Multidisciplinary stakeholders (>1 profession)7
      Steering group members
      Not stated in n = 5.
      Surgeons/doctors9
      Nurses3
      Allied health professionals5
      Patients and care givers3
      Other1
      Multidisciplinary stakeholders (>1 profession)5
      Training / experience in priority setting (e.g., JLA advisor)5
      a Does not total 17 due to international studies.
      b In the original RCS classification, burns are included in the ‘trauma’ subspecialty group.
      c The JLA was established in 2004.
      d Inclusion of question type per study, not per question; does not total 17 as some studies included multiple question types.
      e Unclear in n = 1 study.
      f Not stated in n = 5.
      Table 3Study methodology.
      N
      Methods for gathering uncertainties (per study)
      Systematic review1
      Clinical guidelines and/or policy documents2
      Survey9
      Stakeholder consultation/focus group4
      Methodology of research prioritisation (per study)
      James Lind Alliance5
      Qualitative only3
      Delphi techniques3
      Other survey (single stage)3
      Consensus conference2
      Mixed methods1
      Average number of uncertainties/priorities, per stage (interquartile range)
      Uncertainty gathering
      Unclear or N/A in n = 6.
      41 (13–719)
      Interim priority setting
      Unclear or N/A in n = 10.
      30 (26–30)
      Final consensus meeting
      Unclear or N/A in n = 5.
      10 (9–12)
      Median number of stakeholders per stage (interquartile range)
      Gathering uncertainties
      Unclear or N/A in n = 11.
      197 (79–338)
      Interim priority setting
      Unclear or N/A in n = 9.
      191 (127–289)
      Final prioritisation or consensus meeting
      Unclear or N/A in n = 5.


      One stage prioritisation process
      25 (22–29)

      42 (25–248)
      a Unclear or N/A in n = 6.
      b Unclear or N/A in n = 10.
      c Unclear or N/A in n = 5.
      d Unclear or N/A in n = 11.
      e Unclear or N/A in n = 9.
      f Unclear or N/A in n = 5.
      Table 4Comprehensiveness of reporting (REPRISE).
      ItemReferencesN (%)
      Context and scope
       Define geographical scope11,14,23–29,15–2217 (100%)
       Define health area/focus11,14,23–29,15–2217 (100%)
       Define end-users of the research11,192 (12%)
       Identify the research focus11,14,23–29,15–2217 (100%)
       Identify the type of research question14,15,18,20,21,23,267 (41%)
       Identify the time frame20,252 (12%)
      Governance and team
       Describe the selection of project leaders and team11,14,15,26–297 (41%)
       Describe the characteristics of the project leader and team members14,19,23,284 (24%)
       Describe any prior training or experience in research priority setting14,20,25,264 (24%)
      Inclusion of stakeholders/participants
       Define the inclusion criteria for stakeholder groups11,14,23–29,15–2217 (100%)
       State the strategy for identifying and engaging stakeholders11,14,15,19,20,22,24–29,1713 (76%)
       Indicate the number of participants and/or organisations involved11,14,25–29,15–17,19–2315 (88%)
       Describe the characteristics of stakeholders11,14,26–29,15–17,19–2316 (94%)
       State if reimbursement for participation was provided0
      Identification and collection of research topics/questions
       Identify the approach to priority setting11,14,23–29,15–2217 (100%)
       Describe the methods for collecting research topics or questions11,14,26–29,15–17,19,20,22,24,2514 (82%)
       Describe the framework used to organise/aggregate topics or questions11,14,16,19,20,22,26–29,1711 (65%)
       Describe methods and reason for initial removal of topics or questions14,16,26–285 (29%)
       Describe methods for checking if research questions have already been answered14,25–285 (29%)
       Describe the number of research topics or questions11,14,27–29,15,16,19–22,25,2613 (76%)
      Prioritisation of research topics/questions
       Describe methods for prioritising or achieving consensus11,14,27–29,15,16,19,21–24,2613 (76%)
       Provide reasons for excluding research topics or questions14,16,26–296 (35%)
      Output
       Describe the dissemination of the priority setting exercise14,15,19,22,26–287 (41%)
       Published in a peer-reviewed journal11,14,23,24,26–29,15–2216 (94%)
       Define the specificity of the research priorities14,15,24–29,16–2316 (94%)
      Evaluation and feedback
       Describe how the priority setting exercise was evaluated and any changes that were made14,192 (12%)
       Describe how priorities were made accessible for review by stakeholders141 (6%)
      Translation and implementation
       Outline the strategy or action plan for implementing priorities15,16,19,22,26,276 (35%)
       Describe how the impact will be measured14,16,293 (18%)
      Funding and conflicts of interest
       State sources of funding14,19,20,23,25–27,298 (47%)
       Outline the budget and/or cost of the project0
       Declare any conflicts of interest14,19,20,26–297 (41%)
      Figure 2
      Figure 2Overall completeness of reporting of REPRISE checklist by the year of publication of priority setting exercise.
      Table 5Recommendations for future priority setting exercises in plastic surgery.
      1. Research priority setting exercises focussing on traumatic injuries and (non-breast) cancer reconstruction
      2. Inclusion of steering group member(s) with experience in research priority setting
      3. Multidisciplinary research priority setting initiatives
      4. Increased representation of lower income countries and patient stakeholders
      5. Improved quality of reporting across all domains of REPRISE (in particular, the recruitment and characteristics of steering group members and stakeholders, the processes for collecting and prioritising uncertainties and funding disclosures and conflicts of interest)
      6. Development and widespread use of strategies to assess implementation and impact of priorities

      Results

      Identification of studies

      The database search returned 3899 de-duplicated citations, of which 17 were included in the final analysis (Figure 1). Five publications were merged as they represented one priority setting exercise.
      • Weber B.F.
      • Carrougher G.J.
      • Marvin J.A.
      • Bayley E.W.
      • Knighton J.
      • Rutan R.L.
      Research priorities for burn nursing: nursing recruitment, retention, and other administrative issues.
      • Marvin J.
      • Carrougher G.
      • Bayley E.
      • Knighton J.
      • Rutan R.
      • Weber B.
      Burn nursing Delphi study: pain management.
      • Knighton J.
      • Carrougher G.J.
      • Marvin J.A.
      • Bayley E.W.
      • Rutan R.L.
      • Weber B.
      Research priorities for burn nursing: report of the psychosocial issues group.
      • Carrougher G.J.
      • Marvin J.A.
      • Bayley E.W.
      • Knighton J.
      • Rutan R.L.
      • Weber B.
      Research priorities for burn nursing: report of the wound care and infection control group.
      • Bayley E.W.
      • Carrougher G.J.
      • Marvin J.A.
      • Knighton J.
      • Rutan R.L.
      • Weber B.
      Research priorities for burn nursing: patient, nurse, and burn prevention education.
      Two studies identified from the JLA website were not published in peer-reviewed journals; data were therefore extracted from available JLA reports.

      The ‘top 12’ priorities for research in Cleft Lip and Palate. James Lind Alliance Cleft Lip & Palate Priority Setting Partnership. Available from: https://www.jla.nihr.ac.uk/priority-setting-partnerships/diabetes-type-1/cleft-lip-and-palate/top-10-priorities/.

      ,
      • Madden M.
      Partnership Gives Research Voice to Pressure Ulcer Patients.

      Context and scope

      Most priority setting initiatives were conducted in single country (14/17)
      • Henderson J.
      • Reid A.
      • Jain A.
      Use of a modified BAPRAS Delphi process for research priority setting in Plastic Surgery in the UK.
      ,
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      ,

      The ‘top 12’ priorities for research in Cleft Lip and Palate. James Lind Alliance Cleft Lip & Palate Priority Setting Partnership. Available from: https://www.jla.nihr.ac.uk/priority-setting-partnerships/diabetes-type-1/cleft-lip-and-palate/top-10-priorities/.

      • Ingram J.R.
      • Abbott R.
      • Ghazavi M.
      • et al.
      The hidradenitis suppurativa priority setting partnership.
      • Ford S.
      List of 12 Priorities for Pressure Ulcer Study Identified by UK Researchers.
      • Zhong T.
      • Mahajan A.
      • Cowan K.
      • et al.
      Identifying the top research priorities in postmastectomy breast cancer reconstruction: a James Lind Alliance priority setting partnership.
      ,
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      ,
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      ,
      • Wiechman V.
      • Holavanahalli R.
      Burn survivor focus group.
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      • Jagnoor J.
      • Bekker S.
      • Chamania S.
      • Potokar T.
      • Ivers R.
      Identifying priority policy issues and health system research questions associated with recovery outcomes for burns survivors in India: a qualitative inquiry.
      ,
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.
      Anonymous
      Outcomes measurement in pediatric burn care: an agenda for research: executive summary and final report.
      • Rankin M.
      • Borah G.L.
      • Kosa E.
      Research priorities and concerns of plastic surgical nurses.
      and high-income (16/17)
      • Henderson J.
      • Reid A.
      • Jain A.
      Use of a modified BAPRAS Delphi process for research priority setting in Plastic Surgery in the UK.
      ,
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      ,

      The ‘top 12’ priorities for research in Cleft Lip and Palate. James Lind Alliance Cleft Lip & Palate Priority Setting Partnership. Available from: https://www.jla.nihr.ac.uk/priority-setting-partnerships/diabetes-type-1/cleft-lip-and-palate/top-10-priorities/.

      • Ingram J.R.
      • Abbott R.
      • Ghazavi M.
      • et al.
      The hidradenitis suppurativa priority setting partnership.
      • Ford S.
      List of 12 Priorities for Pressure Ulcer Study Identified by UK Researchers.
      • Zhong T.
      • Mahajan A.
      • Cowan K.
      • et al.
      Identifying the top research priorities in postmastectomy breast cancer reconstruction: a James Lind Alliance priority setting partnership.
      • Sadideen H.
      • Akhavani M.
      • Mosahebi A.
      • Harris P.A.
      Consensus research priorities for “Brazilian Butt Lift” (BBL) in the UK: a BAAPS-led Delphi survey of expert clinicians and researchers.
      ,
      • MacDermid J.C.
      • Fess E.E.
      • Bell-Krotoski J.
      • et al.
      A research agenda for Hand Therapy.
      ,
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      ,
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      ,
      • Wiechman V.
      • Holavanahalli R.
      Burn survivor focus group.
      ,
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      ,
      • Richard R.
      • Baryza M.J.
      • Carr J.A.
      • et al.
      Burn rehabilitation and research: proceedings of a consensus summit.
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.
      Anonymous
      Outcomes measurement in pediatric burn care: an agenda for research: executive summary and final report.
      • Rankin M.
      • Borah G.L.
      • Kosa E.
      Research priorities and concerns of plastic surgical nurses.
      settings (Table 2). The three international studies included only high-income countries (United States, Canada, UK, Australia and Norway)
      • MacDermid J.C.
      • Fess E.E.
      • Bell-Krotoski J.
      • et al.
      A research agenda for Hand Therapy.
      ,
      • Richard R.
      • Baryza M.J.
      • Carr J.A.
      • et al.
      Burn rehabilitation and research: proceedings of a consensus summit.
      ,
      • Sadideen H.
      • Akhavani M.
      • Mosahebi A.
      • Harris P.A.
      Consensus research priorities for “Brazilian Butt Lift” (BBL) in the UK: a BAAPS-led Delphi survey of expert clinicians and researchers.
      Only one study was conducted in a lower-middle income setting (India),
      • Jagnoor J.
      • Bekker S.
      • Chamania S.
      • Potokar T.
      • Ivers R.
      Identifying priority policy issues and health system research questions associated with recovery outcomes for burns survivors in India: a qualitative inquiry.
      and none in low-income settings. More priority setting initiatives focussed on burns (6/17)
      • Jagnoor J.
      • Bekker S.
      • Chamania S.
      • Potokar T.
      • Ivers R.
      Identifying priority policy issues and health system research questions associated with recovery outcomes for burns survivors in India: a qualitative inquiry.
      ,
      • Richard R.
      • Baryza M.J.
      • Carr J.A.
      • et al.
      Burn rehabilitation and research: proceedings of a consensus summit.
      ,
      Anonymous
      Outcomes measurement in pediatric burn care: an agenda for research: executive summary and final report.
      ,
      • Wiechman V.
      • Holavanahalli R.
      Burn survivor focus group.
      ,
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      ,
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.
      and hand and upper limb surgery (4/17),
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      ,
      • MacDermid J.C.
      • Fess E.E.
      • Bell-Krotoski J.
      • et al.
      A research agenda for Hand Therapy.
      than other subspecialty groups. Some burns priority setting initiatives had a specific focus, e.g., paediatric burn care,
      Anonymous
      Outcomes measurement in pediatric burn care: an agenda for research: executive summary and final report.
      burn nursing,
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.
      burn recovery.
      • Jagnoor J.
      • Bekker S.
      • Chamania S.
      • Potokar T.
      • Ivers R.
      Identifying priority policy issues and health system research questions associated with recovery outcomes for burns survivors in India: a qualitative inquiry.
      and rehabilitation,
      • Richard R.
      • Baryza M.J.
      • Carr J.A.
      • et al.
      Burn rehabilitation and research: proceedings of a consensus summit.
      which may account for some repetition of the topic. Similarly, the British Association of Hand Therapists regularly repeat their priority setting exercise,
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      ,
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      hence the duplication of UK hand therapy priority setting. No priority setting initiatives were identified in (non-breast) cancer surgery and trauma subspecialty groups. Most priority setting initiatives (13/17) were published after 2004
      • Henderson J.
      • Reid A.
      • Jain A.
      Use of a modified BAPRAS Delphi process for research priority setting in Plastic Surgery in the UK.
      ,
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      ,
      • Ford S.
      List of 12 Priorities for Pressure Ulcer Study Identified by UK Researchers.
      • Zhong T.
      • Mahajan A.
      • Cowan K.
      • et al.
      Identifying the top research priorities in postmastectomy breast cancer reconstruction: a James Lind Alliance priority setting partnership.
      • Sadideen H.
      • Akhavani M.
      • Mosahebi A.
      • Harris P.A.
      Consensus research priorities for “Brazilian Butt Lift” (BBL) in the UK: a BAAPS-led Delphi survey of expert clinicians and researchers.
      ,
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      ,
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      ,
      • Wiechman V.
      • Holavanahalli R.
      Burn survivor focus group.
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      • Jagnoor J.
      • Bekker S.
      • Chamania S.
      • Potokar T.
      • Ivers R.
      Identifying priority policy issues and health system research questions associated with recovery outcomes for burns survivors in India: a qualitative inquiry.
      • Richard R.
      • Baryza M.J.
      • Carr J.A.
      • et al.
      Burn rehabilitation and research: proceedings of a consensus summit.
      ,

      The ‘top 12’ priorities for research in Cleft Lip and Palate. James Lind Alliance Cleft Lip & Palate Priority Setting Partnership. Available from: https://www.jla.nihr.ac.uk/priority-setting-partnerships/diabetes-type-1/cleft-lip-and-palate/top-10-priorities/.

      ,
      • Ingram J.R.
      • Abbott R.
      • Ghazavi M.
      • et al.
      The hidradenitis suppurativa priority setting partnership.
      All studies included priorities relevant to clinical practice; some additionally looked at the delivery of health services
      • MacDermid J.C.
      • Fess E.E.
      • Bell-Krotoski J.
      • et al.
      A research agenda for Hand Therapy.
      ,
      • Wiechman V.
      • Holavanahalli R.
      Burn survivor focus group.
      ,
      • Richard R.
      • Baryza M.J.
      • Carr J.A.
      • et al.
      Burn rehabilitation and research: proceedings of a consensus summit.
      ,
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.
      and basic science.
      • Henderson J.
      • Reid A.
      • Jain A.
      Use of a modified BAPRAS Delphi process for research priority setting in Plastic Surgery in the UK.
      ,
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      For example, in their priority setting process for general plastic surgery, Henderson et al.
      • Henderson J.
      • Reid A.
      • Jain A.
      Use of a modified BAPRAS Delphi process for research priority setting in Plastic Surgery in the UK.
      included both clinical topics (clinical trials and lymphoedema surgery) and basic science topics (tissue engineering and gene therapy) in the final priorities.

      Stakeholder characteristics

      Less than half of studies included patients (8/17)
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      ,
      • Wiechman V.
      • Holavanahalli R.
      Burn survivor focus group.
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      • Jagnoor J.
      • Bekker S.
      • Chamania S.
      • Potokar T.
      • Ivers R.
      Identifying priority policy issues and health system research questions associated with recovery outcomes for burns survivors in India: a qualitative inquiry.
      ,

      The ‘top 12’ priorities for research in Cleft Lip and Palate. James Lind Alliance Cleft Lip & Palate Priority Setting Partnership. Available from: https://www.jla.nihr.ac.uk/priority-setting-partnerships/diabetes-type-1/cleft-lip-and-palate/top-10-priorities/.

      • Ingram J.R.
      • Abbott R.
      • Ghazavi M.
      • et al.
      The hidradenitis suppurativa priority setting partnership.
      • Ford S.
      List of 12 Priorities for Pressure Ulcer Study Identified by UK Researchers.
      • Zhong T.
      • Mahajan A.
      • Cowan K.
      • et al.
      Identifying the top research priorities in postmastectomy breast cancer reconstruction: a James Lind Alliance priority setting partnership.
      or multi-disciplinary professionals (8/17)
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      ,
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      • Jagnoor J.
      • Bekker S.
      • Chamania S.
      • Potokar T.
      • Ivers R.
      Identifying priority policy issues and health system research questions associated with recovery outcomes for burns survivors in India: a qualitative inquiry.
      • Richard R.
      • Baryza M.J.
      • Carr J.A.
      • et al.
      Burn rehabilitation and research: proceedings of a consensus summit.
      ,
      Anonymous
      Outcomes measurement in pediatric burn care: an agenda for research: executive summary and final report.
      ,

      The ‘top 12’ priorities for research in Cleft Lip and Palate. James Lind Alliance Cleft Lip & Palate Priority Setting Partnership. Available from: https://www.jla.nihr.ac.uk/priority-setting-partnerships/diabetes-type-1/cleft-lip-and-palate/top-10-priorities/.

      ,
      • Ingram J.R.
      • Abbott R.
      • Ghazavi M.
      • et al.
      The hidradenitis suppurativa priority setting partnership.
      ,
      • Zhong T.
      • Mahajan A.
      • Cowan K.
      • et al.
      Identifying the top research priorities in postmastectomy breast cancer reconstruction: a James Lind Alliance priority setting partnership.
      as stakeholders (Table 2). Two studies included nurse only stakeholders,
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.
      ,
      • Rankin M.
      • Borah G.L.
      • Kosa E.
      Research priorities and concerns of plastic surgical nurses.
      three included therapist only stakeholders,
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      • MacDermid J.C.
      • Fess E.E.
      • Bell-Krotoski J.
      • et al.
      A research agenda for Hand Therapy.
      two surgeon only stakeholders
      • Henderson J.
      • Reid A.
      • Jain A.
      Use of a modified BAPRAS Delphi process for research priority setting in Plastic Surgery in the UK.
      ,
      • Sadideen H.
      • Akhavani M.
      • Mosahebi A.
      • Harris P.A.
      Consensus research priorities for “Brazilian Butt Lift” (BBL) in the UK: a BAAPS-led Delphi survey of expert clinicians and researchers.
      and one patient/caregiver only stakeholders.
      • Wiechman V.
      • Holavanahalli R.
      Burn survivor focus group.
      This included priority setting initiatives focussed on surgical nursing.
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.
      ,
      • Rankin M.
      • Borah G.L.
      • Kosa E.
      Research priorities and concerns of plastic surgical nurses.
      and physical therapy.
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      • MacDermid J.C.
      • Fess E.E.
      • Bell-Krotoski J.
      • et al.
      A research agenda for Hand Therapy.
      Five studies reported a gender breakdown of stakeholders.
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      ,
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.
      Anonymous
      Outcomes measurement in pediatric burn care: an agenda for research: executive summary and final report.
      • Rankin M.
      • Borah G.L.
      • Kosa E.
      Research priorities and concerns of plastic surgical nurses.
      ,
      • Zhong T.
      • Mahajan A.
      • Cowan K.
      • et al.
      Identifying the top research priorities in postmastectomy breast cancer reconstruction: a James Lind Alliance priority setting partnership.
      The number of involved stakeholders varied, particularly at the uncertainty gathering and interim prioritisation stages (Table 3). This may reflect how specialised the topic being prioritised is; for example, in their priority setting exercise for ‘Brazilian Butt Lift’ (BBL) Sadideen et al.
      • Sadideen H.
      • Akhavani M.
      • Mosahebi A.
      • Harris P.A.
      Consensus research priorities for “Brazilian Butt Lift” (BBL) in the UK: a BAAPS-led Delphi survey of expert clinicians and researchers.
      only invited surgeons who performed a large volume of fat grafting and BBL surgery (n = 10). Few studies included in their steering groups multidisciplinary professionals (3/17),
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      ,
      • Ingram J.R.
      • Abbott R.
      • Ghazavi M.
      • et al.
      The hidradenitis suppurativa priority setting partnership.
      ,
      • Zhong T.
      • Mahajan A.
      • Cowan K.
      • et al.
      Identifying the top research priorities in postmastectomy breast cancer reconstruction: a James Lind Alliance priority setting partnership.
      patients and caregivers (3/17)
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      ,
      • Ingram J.R.
      • Abbott R.
      • Ghazavi M.
      • et al.
      The hidradenitis suppurativa priority setting partnership.
      ,
      • Zhong T.
      • Mahajan A.
      • Cowan K.
      • et al.
      Identifying the top research priorities in postmastectomy breast cancer reconstruction: a James Lind Alliance priority setting partnership.
      or advisors with training in priority setting (4/17); Table 2.

      Study methodology

      The JLA.
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      ,

      The ‘top 12’ priorities for research in Cleft Lip and Palate. James Lind Alliance Cleft Lip & Palate Priority Setting Partnership. Available from: https://www.jla.nihr.ac.uk/priority-setting-partnerships/diabetes-type-1/cleft-lip-and-palate/top-10-priorities/.

      • Ingram J.R.
      • Abbott R.
      • Ghazavi M.
      • et al.
      The hidradenitis suppurativa priority setting partnership.
      • Ford S.
      List of 12 Priorities for Pressure Ulcer Study Identified by UK Researchers.
      • Zhong T.
      • Mahajan A.
      • Cowan K.
      • et al.
      Identifying the top research priorities in postmastectomy breast cancer reconstruction: a James Lind Alliance priority setting partnership.
      and qualitative approaches.
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      ,
      • Wiechman V.
      • Holavanahalli R.
      Burn survivor focus group.
      ,
      • Jagnoor J.
      • Bekker S.
      • Chamania S.
      • Potokar T.
      • Ivers R.
      Identifying priority policy issues and health system research questions associated with recovery outcomes for burns survivors in India: a qualitative inquiry.
      ,
      • Richard R.
      • Baryza M.J.
      • Carr J.A.
      • et al.
      Burn rehabilitation and research: proceedings of a consensus summit.
      ,
      Anonymous
      Outcomes measurement in pediatric burn care: an agenda for research: executive summary and final report.
      were the most common methods of prioritisation (both 5/17), followed by Delphi techniques,
      • Henderson J.
      • Reid A.
      • Jain A.
      Use of a modified BAPRAS Delphi process for research priority setting in Plastic Surgery in the UK.
      ,
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.
      ,
      • Sadideen H.
      • Akhavani M.
      • Mosahebi A.
      • Harris P.A.
      Consensus research priorities for “Brazilian Butt Lift” (BBL) in the UK: a BAAPS-led Delphi survey of expert clinicians and researchers.
      other surveys.
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      ,
      • Rankin M.
      • Borah G.L.
      • Kosa E.
      Research priorities and concerns of plastic surgical nurses.
      ,
      • MacDermid J.C.
      • Fess E.E.
      • Bell-Krotoski J.
      • et al.
      A research agenda for Hand Therapy.
      (both 3/17) or mixed methods (1/17)
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      The number of priorities varied at different stages of prioritisation, particularly at the uncertainty gathering stage (Table 3). The median number of final priorities was 10 (interquartile range: 9–12), but one study had as many as 101.
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.

      Quality of reporting

      The quality of reporting varied across studies. Out of a possible 32 items included in the modified REPRISE checklist (Table 4), the number of items met ranged from 9 (28%) to 27 (84%). The overall completeness of reporting, as defined by the number of checklist items met, did not correlate significantly with the year of publication (r = 0.43, p = 0.084). All 17 studies defined the geographical scope, health area or focus, and research focus of the priority setting exercise. Basic characteristics of stakeholders were described in most studies (16/17),
      • Henderson J.
      • Reid A.
      • Jain A.
      Use of a modified BAPRAS Delphi process for research priority setting in Plastic Surgery in the UK.
      ,
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      ,
      • Rankin M.
      • Borah G.L.
      • Kosa E.
      Research priorities and concerns of plastic surgical nurses.

      The ‘top 12’ priorities for research in Cleft Lip and Palate. James Lind Alliance Cleft Lip & Palate Priority Setting Partnership. Available from: https://www.jla.nihr.ac.uk/priority-setting-partnerships/diabetes-type-1/cleft-lip-and-palate/top-10-priorities/.

      • Ingram J.R.
      • Abbott R.
      • Ghazavi M.
      • et al.
      The hidradenitis suppurativa priority setting partnership.
      ,
      • Zhong T.
      • Mahajan A.
      • Cowan K.
      • et al.
      Identifying the top research priorities in postmastectomy breast cancer reconstruction: a James Lind Alliance priority setting partnership.
      ,
      • Sadideen H.
      • Akhavani M.
      • Mosahebi A.
      • Harris P.A.
      Consensus research priorities for “Brazilian Butt Lift” (BBL) in the UK: a BAAPS-led Delphi survey of expert clinicians and researchers.
      ,
      • MacDermid J.C.
      • Fess E.E.
      • Bell-Krotoski J.
      • et al.
      A research agenda for Hand Therapy.
      ,
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      ,
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      ,
      • Wiechman V.
      • Holavanahalli R.
      Burn survivor focus group.
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      • Jagnoor J.
      • Bekker S.
      • Chamania S.
      • Potokar T.
      • Ivers R.
      Identifying priority policy issues and health system research questions associated with recovery outcomes for burns survivors in India: a qualitative inquiry.
      • Richard R.
      • Baryza M.J.
      • Carr J.A.
      • et al.
      Burn rehabilitation and research: proceedings of a consensus summit.
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.
      Anonymous
      Outcomes measurement in pediatric burn care: an agenda for research: executive summary and final report.
      but these were often limited to occupation and did not specify age, gender or income status. Few studies identified the project leaders or steering group members (6/17),
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      ,
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      ,
      • Ingram J.R.
      • Abbott R.
      • Ghazavi M.
      • et al.
      The hidradenitis suppurativa priority setting partnership.
      ,
      • Zhong T.
      • Mahajan A.
      • Cowan K.
      • et al.
      Identifying the top research priorities in postmastectomy breast cancer reconstruction: a James Lind Alliance priority setting partnership.
      described their characteristics (a minimum of total number, stakeholder type and occupation; 4/17), or described how the individuals within these groups were selected (7/17)
      • Henderson J.
      • Reid A.
      • Jain A.
      Use of a modified BAPRAS Delphi process for research priority setting in Plastic Surgery in the UK.
      ,
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      ,
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      ,
      • Ingram J.R.
      • Abbott R.
      • Ghazavi M.
      • et al.
      The hidradenitis suppurativa priority setting partnership.
      • Ford S.
      List of 12 Priorities for Pressure Ulcer Study Identified by UK Researchers.
      • Zhong T.
      • Mahajan A.
      • Cowan K.
      • et al.
      Identifying the top research priorities in postmastectomy breast cancer reconstruction: a James Lind Alliance priority setting partnership.
      • Sadideen H.
      • Akhavani M.
      • Mosahebi A.
      • Harris P.A.
      Consensus research priorities for “Brazilian Butt Lift” (BBL) in the UK: a BAAPS-led Delphi survey of expert clinicians and researchers.
      Very few studies reported how the final priorities were made available for review by stakeholders (1/17),
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      or how the completed exercise was evaluated (2/17)
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      ,
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      Few reported a strategy for implementing the research priorities (6/17)
      • Henderson J.
      • Reid A.
      • Jain A.
      Use of a modified BAPRAS Delphi process for research priority setting in Plastic Surgery in the UK.
      ,
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      ,
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      ,
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      ,
      • Ingram J.R.
      • Abbott R.
      • Ghazavi M.
      • et al.
      The hidradenitis suppurativa priority setting partnership.
      ,
      • Ford S.
      List of 12 Priorities for Pressure Ulcer Study Identified by UK Researchers.
      or measuring their impact (2/17)
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      ,
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      Most strategies for implementation described liaison with funding bodies (professional organisations, e.g., BAPRAS and national funding bodies, e.g., NIHR) to inform a programme of research. Two studies explicitly stated the amount of funding achieved for proposals incorporating their research priorities. Karantana et al. conducted an ‘impact’ survey of hand clinicians, demonstrating £3 million of competitively awarded funding achieved by researchers who used the research priorities for hand and wrist conditions to strengthen their applications. Steward et al. were able to provide funding for a 60-credit MSc research module and provide a research grant (£5000 per annum for 3 years) for proposals responding to their research priorities for hand therapy, via the British Association of Hand Therapy. No studies reported if reimbursement was provided for patient participation, and no studies outlined the budget for the project.

      Discussion

      This systematic review has comprehensively summarised and appraised global research priority setting initiatives in plastic surgery. Overall, the findings demonstrate underrepresentation of stakeholders (particularly patients) from lower-income countries, as well as lack of multidisciplinary initiatives. Compliance with reporting standards for research priority setting was variable and especially deficient in the areas of governance and team members, evaluation and feedback, translation and implementation, and funding and conflicts of interest.
      The scope of the priority setting initiatives varied widely; some focussed on individual conditions or procedures,

      The ‘top 12’ priorities for research in Cleft Lip and Palate. James Lind Alliance Cleft Lip & Palate Priority Setting Partnership. Available from: https://www.jla.nihr.ac.uk/priority-setting-partnerships/diabetes-type-1/cleft-lip-and-palate/top-10-priorities/.

      • Ingram J.R.
      • Abbott R.
      • Ghazavi M.
      • et al.
      The hidradenitis suppurativa priority setting partnership.
      • Ford S.
      List of 12 Priorities for Pressure Ulcer Study Identified by UK Researchers.
      • Zhong T.
      • Mahajan A.
      • Cowan K.
      • et al.
      Identifying the top research priorities in postmastectomy breast cancer reconstruction: a James Lind Alliance priority setting partnership.
      • Sadideen H.
      • Akhavani M.
      • Mosahebi A.
      • Harris P.A.
      Consensus research priorities for “Brazilian Butt Lift” (BBL) in the UK: a BAAPS-led Delphi survey of expert clinicians and researchers.
      whilst others focussed on the subspecialties
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      ,
      • Wiechman V.
      • Holavanahalli R.
      Burn survivor focus group.
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      • Jagnoor J.
      • Bekker S.
      • Chamania S.
      • Potokar T.
      • Ivers R.
      Identifying priority policy issues and health system research questions associated with recovery outcomes for burns survivors in India: a qualitative inquiry.
      • Richard R.
      • Baryza M.J.
      • Carr J.A.
      • et al.
      Burn rehabilitation and research: proceedings of a consensus summit.
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.
      Anonymous
      Outcomes measurement in pediatric burn care: an agenda for research: executive summary and final report.
      ,
      • MacDermid J.C.
      • Fess E.E.
      • Bell-Krotoski J.
      • et al.
      A research agenda for Hand Therapy.
      or plastic surgery overall.
      • Henderson J.
      • Reid A.
      • Jain A.
      Use of a modified BAPRAS Delphi process for research priority setting in Plastic Surgery in the UK.
      ,
      • Rankin M.
      • Borah G.L.
      • Kosa E.
      Research priorities and concerns of plastic surgical nurses.
      There were several priority setting initiatives for burns
      • Jagnoor J.
      • Bekker S.
      • Chamania S.
      • Potokar T.
      • Ivers R.
      Identifying priority policy issues and health system research questions associated with recovery outcomes for burns survivors in India: a qualitative inquiry.
      ,
      • Richard R.
      • Baryza M.J.
      • Carr J.A.
      • et al.
      Burn rehabilitation and research: proceedings of a consensus summit.
      ,
      Anonymous
      Outcomes measurement in pediatric burn care: an agenda for research: executive summary and final report.
      ,
      • Wiechman V.
      • Holavanahalli R.
      Burn survivor focus group.
      ,
      • Broerse J.E.W.
      • Zweekhorst M.B.M.
      • van Rensen A.J.M.L.
      • de Haan M.J.M.
      Involving burn survivors in agenda setting on burn research: an added value?.
      ,
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.
      and hand conditions;
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      ,
      • MacDermid J.C.
      • Fess E.E.
      • Bell-Krotoski J.
      • et al.
      A research agenda for Hand Therapy.
      these tended to have more non-multidisciplinary steering groups and focussed on the care delivered by specific professions (e.g., nursing).
      • Marvin J.A.
      • Carrougher G.
      • Bayley B.
      • Weber B.
      • Knighton J.
      • Rutan R.
      Burn nursing Delphi study. Setting research priorities.
      or therapy.
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      ,
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      ,
      • Richard R.
      • Baryza M.J.
      • Carr J.A.
      • et al.
      Burn rehabilitation and research: proceedings of a consensus summit.
      ,
      • MacDermid J.C.
      • Fess E.E.
      • Bell-Krotoski J.
      • et al.
      A research agenda for Hand Therapy.
      Arguably, a better approach would be to work cohesively with multidisciplinary input to maximise clinical relevance of the final priorities and reduce repetition, particularly with respect to subsequent funding applications. No priority setting initiatives were identified in the trauma and non-breast cancer categories, though major trauma and skin cancer JLA priority setting partnerships are in progress. Some studies, e.g., Henderson et al.
      • Henderson J.
      • Reid A.
      • Jain A.
      Use of a modified BAPRAS Delphi process for research priority setting in Plastic Surgery in the UK.
      had a broad scope traversing clinical practice and basic science; however, the distinct funding streams for clinical and translational research would favour a more clearly defined scope.
      With regards to stakeholder characteristics, there was only one priority setting exercise from a lower middle-income country.
      • Jagnoor J.
      • Bekker S.
      • Chamania S.
      • Potokar T.
      • Ivers R.
      Identifying priority policy issues and health system research questions associated with recovery outcomes for burns survivors in India: a qualitative inquiry.
      , and none from low-income countries, despite the burden of disease (especially, burns and trauma) in lower income nations.
      • Rybarczyk M.M.
      • Schafer J.M.
      • Elm C.M.
      • et al.
      A systematic review of burn injuries in low- and middle-income countries: epidemiology in the WHO-defined African Region.
      ,
      • Whitaker J.
      • O'Donohoe N.
      • Denning M.
      • et al.
      Assessing trauma care systems in low-income and middle-income countries: a systematic review and evidence synthesis mapping the Three Delays framework to injury health system assessments.
      Increasing the participation of stakeholders from lower-income countries will increase generalisability of the final priorities but raises questions for study design. For example, how to define adequate international representation and how to manage international data, particularly when local factors (e.g., health infrastructure and sociocultural beliefs) may significantly affect prioritisation behaviours.
      • Jagnoor J.
      • Bekker S.
      • Chamania S.
      • Potokar T.
      • Ivers R.
      Identifying priority policy issues and health system research questions associated with recovery outcomes for burns survivors in India: a qualitative inquiry.
      ,
      • Lee A.
      • Davies A.
      • Young A.E.
      Systematic review of international Delphi surveys for core outcome set development: representation of international patients.
      Patients were underrepresented as both steering group members and stakeholders, despite clear differences in their prioritisation behaviours compared with healthcare professionals.
      • Chalmers I.
      • Bracken M.B.
      • Djulbegovic B.
      • et al.
      How to increase value and reduce waste when research priorities are set.
      Research has shown that patients are less likely to prioritise pharmacological and surgical interventions (the focus of most registered trials), favouring education and training, service delivery, and physical and psychological interventions.
      • Chalmers I.
      • Bracken M.B.
      • Djulbegovic B.
      • et al.
      How to increase value and reduce waste when research priorities are set.
      Neglecting patients’ needs is a common cause of research waste and should be avoided through adequate representation at steering group and stakeholder levels. Although basic stakeholder characteristics were described in most studies, the level of detail varied and often did not include age, gender or ethnicity, as recommended by the World Health Organisation.
      World Health Organization
      Sufficient demographic information is essential for fully informed interpretation of the final priorities. For example, Karantana et al. in their priority setting exercise were aiming to identify unanswered questions about the management of common conditions affecting adult hands and wrists. The patient and carer stakeholders who participated in the prioritisation exercise were mainly older females, which probably biased the final priorities away from traumatic hand injuries.
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      There is no consensus on appropriate methods of prioritisation, stakeholder sample sizes or the numbers of final priorities, which is reflected in the variability of published studies. Although published guidance exists (e.g., REPRISE),
      • Tong A.
      • Synnot A.
      • Crowe S.
      • et al.
      Reporting guideline for priority setting of health research (REPRISE).
      there are no universally recommended quality assessment tools for studies of research prioritisation, resulting in poor quality reporting across various domains. Few studies have described the recruitment and characteristics of steering group members, their conflicts of interest or funding sources, despite the potential for bias. Furthermore, only JLA-associated initiatives included an advisor experienced in priority setting methodology. A minority of studies explicitly described reasons for exclusion of research topics. A flow chart of uncertainties, clearly depicting the addition and removal of topics, such as that used by Karantana et al.,
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      provides clarity and reproducibility. Many studies did not report cross-checking uncertainties with published systematic reviews, risking unjustified (and sometimes harmful) duplication of research,
      • Chalmers I.
      • Bracken M.B.
      • Djulbegovic B.
      • et al.
      How to increase value and reduce waste when research priorities are set.
      , another cause of research waste. No studies reported patient reimbursement, though this is considered best practice.
      Working with our patient and public voices (PPV) partners. Reimbursing Expenses and Paying Involvement Payments.
      The main aim of research priority setting is to streamline funding for research questions deemed most important by key stakeholders.
      • Staley K.
      • Crowe S.
      • Crocker J.C.
      • Madden M.
      • Greenhalgh T.
      What happens after James Lind Alliance Priority Setting Partnerships? A qualitative study of contexts, processes and impacts.
      Despite this, few studies have described a strategy for implementation of priorities or measuring their impact. Early planning and resourcing for dissemination of the final priorities should be encouraged.
      • Staley K.
      • Crowe S.
      • Crocker J.C.
      • Madden M.
      • Greenhalgh T.
      What happens after James Lind Alliance Priority Setting Partnerships? A qualitative study of contexts, processes and impacts.
      For example, the Dutch Burns Foundation compared top-ranked priorities with their organisation's current research funding, and a pilot programme was developed to address under-funded areas. Greater awareness and training regarding research priority setting amongst funders and academic journals are also required.
      • Staley K.
      • Crowe S.
      • Crocker J.C.
      • Madden M.
      • Greenhalgh T.
      What happens after James Lind Alliance Priority Setting Partnerships? A qualitative study of contexts, processes and impacts.
      For example, the National Institute for Health Research in the UK now fund an annual call for studies addressing JLA research priorities.

      NIHR. NIHR James Lind Alliance Priority Setting Partnerships rolling call. Available from: https://www.nihr.ac.uk/documents/nihr-james-lind-alliance-priority-setting-partnerships-rolling-call/28569.

      The British Association of Hand Therapy repeats their priority setting exercise regularly, allowing them to identify both persistently highly ranked research topics which require ongoing investment (e.g., complex regional pain syndrome), as well as emerging topics (e.g., treatment modalities)
      • Fournier K.
      Research priorities in hand therapy: report of the 2008 UK membership survey.
      ,
      • Steward B.
      The BAHT 2004 R&D prioritisation exercise: results of a study using a nominal group technique to identify priority research topics and research training needs for hand therapists.
      Regularly repeating priority setting exercises could be a useful method for assessing the implementation and impact of priorities, but further consideration is needed to determine appropriate time intervals on a case-by-case basis. Future researchers could also consider conducting an ‘impact survey’ to determine how much competitive funding was awarded to proposals referencing the research priority exercise.
      • Karantana A.
      • Davis T.
      • Kennedy D.
      • et al.
      Common hand and wrist conditions: creation of UK research priorities defined by a James Lind Alliance Priority Setting Partnership.
      Strengths of this review include the comprehensive search of three electronic databases and hand-searching the JLA repository, with dual-author screening and data extraction. Limitations include lack of non-English language articles and formal risk of bias assessment, although no specific tool exists for research priority setting exercises, and reporting quality was assessed with the REPRISE checklist. Study compliance with some of the REPRISE checklist items was subjective. For example, most studies described stakeholder characteristics, but the level of detail (not specified in REPRISE) varied substantially from only reporting stakeholder type (i.e., patient versus healthcare professional) to detailed demographics (age, gender and ethnicity).

      Conclusion

      Research priority setting exercises have been conducted in most subspecialty areas of plastic and reconstructive surgery, except trauma and non-breast cancer. Reporting quality was variable and generally poor when describing project team and stakeholder characteristics, reasoning for exclusion of research uncertainties, authors’ conflicts of interest and funding sources. A core aim of establishing research priorities is to influence funding allocation, yet strategies to assess their implementation were rarely described. Future initiatives should also consider recruitment of multidisciplinary steering groups, including an advisor with experience in research priority setting, and more patients and stakeholders from lower-income countries.

      Conflicts of Interest

      None declared.

      Financial disclosure statement

      This article presents independent research funded by the National Institute for Health Research (NIHR) Advanced Research Fellowship NIHR 301362. The views expressed are those of the authors and not necessarily those of the NHS, the NIHR or the Department of Health. The study was also supported by the NIHR Biomedical Research Centre at the University Hospitals Bristol and Weston NHS Foundation Trust and the University of Bristol.

      Ethical approval

      Not required.

      Appendix. Supplementary materials

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