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Abstract
Background The General and Adolescent Paediatric Research Network in the UK and Ireland (GAPRUKI) was established in 2016. The aims of GAPRUKI are to unite general paediatricians around the UK and Ireland, to develop research ideas and protocols, and facilitate delivery of multicentre research.
Objectives To undertake a research prioritisation exercise among UK and Ireland general paediatricians.
Methods This was a four-phase study using a modified Delphi survey. The first phase asked for suggested research priorities. The second phase developed ideas and ranked them in priority. In the third phase, priorities were refined; and the final stage used the Hanlon Prioritisation Process to agree on the highest priorities.
Results In phase one, there were 250 questions submitted by 61 GAPRUKI members (66% of the whole membership). For phase two, 92 priorities were scored by 62 members and the mean Likert scale (1–7) scores ranged from 3.13 to 5.77. In a face-to-face meeting (phases three and four), 17 research questions were identified and ultimately 14 priorities were identified and ranked. The four priorities with the highest ranking focused on these three respiratory conditions: asthma, bronchiolitis and acute wheeze. Other priorities were in the diagnosis or management of constipation, urinary tract infection, fever, gastro-oesophageal reflux and also new models of care for scheduled general paediatric clinics.
Conclusion Research priorities for child health in the UK and Ireland have been identified using a robust methodology. The next steps are for studies to be designed and funded to address these priorities.
- child health
- paediatrics
- health services research
- child health services
Data availability statement
Data sharing not applicable as no datasets generated and/or analysed for this study. No datasets generated and/or analysed for this study.
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What is already known on this topic?
Paediatric Emergency Research Network UK and Ireland and other international paediatric research networks have established research priorities of their members.
There are no general paediatric research priorities for the UK.
What this study adds?
General paediatric research priorities for UK and Ireland have been determined using a robust methodology.
These results could be considered by research funders in the UK and Ireland when creating their research strategies.
Introduction
General paediatrics is an underfunded and under-represented area of paediatric research.1 Half of the medicines used for in general medical care of children and infants are given ‘off label’,2 3 and most practice is not evidence based. Commissioned calls for research funding in general paediatrics are unusual and most funding opportunities are for research in paediatric subspecialty areas.1
The lack of available funding presents a challenge to converting research ideas into evidence-based practice.4 The present situation means that 80% of consultant paediatricians have no time in their job plan for research.3 4 Although 47% of newly appointed consultant would like to undertake more research work, only 23% expected to do so in 2017.4
The General and Adolescent Paediatric Research Network in the UK and Ireland (GAPRUKI) was established in 2016 to facilitate research in general paediatrics.5 The GAPRUKI collaborators include general paediatric consultants, trainees, nurses and research personnel in the UK and Ireland who work in district general or tertiary paediatric hospitals. GAPRUKI will identify and develop research ideas and support multicentre studies. GAPRUKI works closely with established networks including the National Institute for Health Research (NIHR) Clinical Research Network (CRN) and General Paediatric Clinical Studies Group.
Other research networks have conducted prioritisation surveys to help plan the future direction of their research; for example, Paediatric Emergency Research Network in the UK and Ireland (PERUKI),6 Paediatric Research in Emergency Departments International Collaborative Network in Australia and New Zealand (PREDICT),7 and other research networks in North America, West Europe and Australia.8 9 Here we report the GAPRUKI general paediatric research priority exercise.
Methods
A four-phase study was conducted using a modified Delphi technique survey10 and the Hanlon Prioritisation Process (HPP) (see figure 1).11 Our survey was distributed using collaborative professional networks.
Prioritisation process.
Stage one: Delphi survey one
Delphi survey one was carried out using the Bristol Online survey tool (21 February–30 March 2018).10 All GAPRUKI members were asked: ‘Thinking about your practice in the field of general paediatrics, both acute and in outpatients, what are the important research questions that need addressing?’ Participants could list up to 10 priorities/questions and were encouraged to submit these in a PICO (Population, Intervention, Control and Outcome) format. Responses were anonymised and response to the survey was taken as implied consent. Submitted questions were reviewed, combining matching themes, and duplicates were removed ahead of stage two.
Stage two: Delphi survey two
Delphi survey two ranked the questions from stage one (7 September–7 November 2018). All GAPRUKI members were asked: ‘Thinking about your practice in the field of general paediatrics, both acute and in outpatients, can you rank these questions according to your consideration for prioritisation of future general paediatric research?’ Ranking was performed by respondents using a Likert scale of 1–7 (1=not a priority, 2=low priority, 3=somewhat priority, 4=neutral, 5=moderate priority, 6=high priority, 7=essential priority).
Stage three: refinement of research questions
Research questions from stage two were reviewed (May–September 2019). GAPRUKI members were invited to attend a face-to-face meeting to refine the research questions in preparation for the HPP. Questions from stage two were allocated to attendees to review the literature and feed back their findings at the meeting in relation to the following criteria7:
Is the question amenable to multicentre research? Questions that were felt to only be relevant to a single centre were excluded as GAPRUKI is a multicentre research network.
Is the question amenable to the PICO format? Questions felt to not be amenable to the PICO format were excluded.
Is there current existing evidence to answer the question? Questions where there was already felt to be an answer in the current literature or a study ongoing at the present time to answer the question were excluded.
To ensure patient and public involvement, invitations were sent to three organisations that represent parents, children and young people (CYP) within healthcare settings (Generation R, Wellchild, National Network of Parent and Carers Forums).
Stage four: the HPP
In the fourth stage (September 2019), following face-to-face review and discussion, members present voted (‘Yes’ or ‘No’ to the following statements) on which questions to take into the final stage of the HPP:
Yes–the question is in (or amenable to) a PICO format and currently there is no answer in the literature or study ongoing to answer the question.
No–the question is either not amenable to a PICO format or there is an answer to the question already in the literature or a study ongoing that will answer the question.
Questions achieving >50% ‘yes’ vote from attendees were included in the final stage of the HPP. Attendees scored each question according to the guidance in table 1. For every question, a mean score in each domain (A, B and C) was calculated and used to determine the HPP priority score and final ranking (HPP=(A+2B)×C).11 Scoring and analysis from the HPP was completed in January 2020.
Hanlon scoring guidance11
Results
Response rates and characteristics of respondents
GAPRUKI had 92 members at stage one and 103 at stage two. There were 61 (66%) responses to the first survey and 62 (60%) to the second survey (figure 1). Twenty-four individuals attended the final HPP meeting, comprising 23 GAPRUKI members, and 1 of the 3 invited organisations representing parents and CYP.
Stage one: Delphi survey one
Two hundred fifty questions were submitted by respondents in survey one (range 3–10 per member). Initial questions were reviewed, combining matching themes and removing duplicates. Ninety-two questions, of different categories, were submitted for survey two as shown in table 2.
Number of questions included in Delphi survey two by category
Stage two: Delphi survey two
Ninety-two questions were ranked in survey two. The mean scores for questions ranged from 3.13 to 5.77 (Likert scale of 1–7: 1=not a priority, 7=essential priority). The top 10 questions are presented in table 3.
Top 10 questions following Delphi survey two
Stage three: refinement of research questions
Following survey two, 92 questions were reviewed. In September 2019, 23 GAPRUKI members attended the face-to-face meeting to present their findings alongside 1 of the 3 invited organisations representing parents and CYP.
Stage four: the HPP
Seventy-five questions were excluded due to not being amenable to a PICO format or multicentre research, there already being an answer in the literature or a study is currently attempting to answer the question. These are listed in the online supplemental material. Of the remaining 17 questions, 3 covered constipation clinic models and 2 focused on urinary tract infection (UTI) sampling methods and were therefore combined, resulting in 14 questions/topics being submitted for the Hanlon scoring process.
Supplemental material
Table 4 shows the final scores and ranking for the HPP alongside the ranking after stage two of the Delphi process for comparison.
Final Hanlon scores and ranking alongside Delphi ranking
Discussion
This is the first study to consider the research priorities for general paediatrics in the UK and Ireland. The range of priority (as evidenced by the Hanlon score) was wide, but respiratory themes occupied the top four ranked priorities. The priorities described here may be useful to funding bodies when considering their funding strategies.
There are inconsistencies across the UK for care of unscheduled respiratory presentations in children12–15 and research may address these atlases of variation, for example, evaluating the impact of pathways of care to standardise practice. Bronchiolitis causes a large burden of disease and use of high-flow nasal cannula (HFNC) therapy for respiratory support has become widely practised, yet evidence for this practice is uncertain.16 17 Respiratory conditions remain common causes for hospitalisation in the UK and interventions are required to safely reduce admissions.18
Some priorities have been the focus of research, for example, HFNC in bronchiolitis16 17 and fluids in sepsis (FISH trial),19 which found that a larger scale trial would not be feasible in the UK population due to lower severity of illness. Indeed, the UK resus council-updated guidance now recommends 10 mL/kg bolus in all situations.20 New methodologies may be required to address priorities where uncertainty regarding best evidence remains. There are currently no studies on the NIHR portfolio that address any of the priority areas, although studies may be underway in other countries.
A number of the research priorities reported are similar to those published by other research networks in the UK, North America, Australia, New Zealand and West Europe.6–9 These comprise respiratory illness, including bronchiolitis and asthma, and also point-of-care testing for febrile infants. Intravenous therapy in asthma was the top GAPRUKI priority and also ranked first in the PREDICT prioritisation exercise,7 while the PERUKI prioritisation ranked this fourth.6
Collaboration with other established networks to design and deliver studies to meet research priorities will be key, including the NIHR CRN and PERUKI but also, if required, internationally with other general paediatric research networks in Canada (Paediatric Inpatient Research Network Canada)21 and Australia (Children’s Inpatient Research Collaboration of Australia and New Zealand).22 Partnerships have already proven successful with GAPRUKI and PERUKI delivering joint projects involving defining significant childhood illness and community-acquired pneumonia.23 24
Other common conditions in the priority list included constipation, UTIs and bedside tests in febrile infants. Patients with these conditions use National Health Service (NHS) resources, yet there exists a large variation in practice both around the UK and within departments.25 Effective management of bladder and bowel problems is important to parents and these conditions impact on quality of life for CYP,26 yet they are underlooked in research at present; and like many paediatric conditions managed every day in primary and secondary care, they still require a more robust evidence base.
This study has a number of strengths. The Delphi process response rates were over 60% and participants were from multiple disciplines in several nations, indicating that our results are widely generalisable. The combination of the Delphi and Hanlon processes was rigorous and yielded a robust final list of priorities. The limitations of this study include the subjective nature of the process and the addition of new members between the first two stages. While the number of GAPRUKI members taking part in stages one and two was very similar, we do not know if these were mostly the same individuals, and introduction of new members to stage two may have led to different ranking of questions compared with if the same members suggested and ranked questions. Ideally, the process would have been completed in under 12 months, but we do not believe that the time between completion of the different stages has affected the relevance of the priorities. Patient and parent/carer input was limited by the response to invitations to join the process. Future research prioritisation initiatives need to explore how to better engage with patient groups.
The COVID-19 pandemic started a few months after the face-to-face meeting, so this process could not consider COVID-19 research priorities. Many COVID-19 priorities, for example, long COVID-19, Paediatric multisystem inflammatory syndrome temporally associated with COVID-19 (PIMS-TS) and vaccination, have already received research investment.
This study has identified research priorities for general paediatricians in the UK and Ireland, which remain, despite the COVID-19 pandemic. On review of the NIHR portfolio (October 2021), we can confirm that there is no funded research in the priority areas identified by GAPRUKI currently being conducted in the UK, although research overseas may be underway. We suggest that funding bodies could seek applications from research teams to undertake clinical trials in the priority areas identified.
Data availability statement
Data sharing not applicable as no datasets generated and/or analysed for this study. No datasets generated and/or analysed for this study.
Ethics statements
Patient consent for publication
Ethics approval
Ethical approval was not required according to the Health Research Authority (HRA) framework decision tool.27
Acknowledgments
All GAPRUKI members who contributed to the prioritisation process.
References
Supplementary materials
Supplementary Data
This web only file has been produced by the BMJ Publishing Group from an electronic file supplied by the author(s) and has not been edited for content.
Supplementary Data
This web only file has been produced by the BMJ Publishing Group from an electronic file supplied by the author(s) and has not been edited for content.
Footnotes
Twitter @katrinacathie, @DrDaveCoghlan
Correction notice This paper has been amended since it was first published. The first affiliation has been updated.
Collaborators On behalf of the GAPRUKI Network.
Contributors CP and AGS designed the Delphi survey. CP compiled the Delphi data. KC managed the Hanlon Prioritisation Process (HPP) and compiled the HPP data. KC wrote the paper, CP revised it. AGS, SB, DC and SWT reviewed and contributed to the manuscript. KC is responsible for the overall content as guarantor.
Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Competing interests None declared.
Provenance and peer review Not commissioned; externally peer reviewed.
Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.