Abstract
In 2017, the British Society for Children’s Orthopaedic Surgery engaged the profession and all relevant stakeholders in two formal research prioritization processes. In this editorial, we describe the impact of this prioritization on funding, and how research in children’s orthopaedics, which was until very recently a largely unfunded and under-investigated area, is now flourishing. Establishing research priorities was a crucial step in this process.
Cite this article: Bone Joint J 2024;106-B(5):422–424.
Research prioritization is increasingly important in orthopaedic surgery, with many of the specialist societies engaging in priority-setting exercises. The processes of generating these priorities are themselves academic activities, involving consensus-building techniques and the engagement of many stakeholders and patients.1 However, like all research, the activity is only worthwhile if it generates impact through change. In the case of research priorities, this change may be demonstrated from those who provide funding, and/or by identifying appropriate priorities from the research community.
Children’s orthopaedics has traditionally been practised empirically, with little evidence to support practice. However, in 2017, the British Society for Children’s Orthopaedic Surgery (BSCOS) began their quest to improve the quality of the evidence that informs practice. Their first step was to define research priorities, but was it worth it?
Establishing research priorities
In order to engage the profession and all relevant stakeholders, BSCOS sought consensus among its members and undertook a formal research prioritization process through the James Lind Alliance (JLA),2 which involved a wide group of professionals and patients.
The initial process involved only orthopaedic surgeons, from both BSCOS and the Orthopaedic Trauma Society (OTS).3 This was a modified Delphi study, overseen by the BSCOS Research committee. The process was simple to administer and quick to achieve results. Uncertainties were prioritized in order of their importance. A total of 86 surgeons contributed to both rounds of the Delphi process, scoring priorities from one (low priority) to five (high priority). Elective topics were ranked higher than those relating to trauma. The top ten elective and top five trauma priorities were identified. The three topics that ranked highest were related to the treatment of slipped capital femoral epiphysis, Perthes’ disease, and infection in bone. Most focused on elective lower limb surgery.
Next, a more formal research prioritization process, under the auspices of the JLA, focused on elective lower limb elective surgery.4 This offered the opportunity for patients and members of the public to have an equal voice to that of clinicians and research workers in influencing the agenda. The JLA’s infrastructure is funded by the National Institute for Health and Care Research (NIHR),5 which oversees the process in a transparent and structured manner. The NIHR encourages active involvement of the public and supports the JLA in feeding back research priorities to national funding bodies.
The JLA process took 18 months between July 2017 and January 2019 to complete. During the first national survey, 388 people generated 1,023 questions, 801 of which were classified as true uncertainties. Following the JLA methodology, 75 uncertainties were developed from the initial 801 questions, and 26 were prioritized through a second national survey and taken to a face-to-face workshop, when the top ten were selected. These included questions dealing with cerebral palsy, common conditions of the hip such as Perthes’ disease and developmental dysplasia of the hip, as well as rehabilitation techniques and methods to improve shared decision-making between clinicians and patients and their families.
Impact on funding
We followed up on the projects in children’s orthopaedics that were funded in the UK, after completion of these research prioritization projects (Table I).
Table I.
Paediatric orthopaedic research projects prioritized by either the James Lind Alliance or the British Society for Children’s Orthopaedic Surgery, which have already been funded.
| Research prioritization | Subject | Funding body |
|---|---|---|
| JLA no. 1 | Core outcomes in children’s lower limb surgery | NIHR doctoral fellowship started 2023 |
| JLA no. 3 and BSCOS elective no. 5 | Lower limb orthopaedic surgery in children with cerebral palsy who can walk | Action Medical Research; recruitment completed, results in Spring 2024 |
| JLA no. 4 | Surgery vs non surgical treatment for Perthes’ disease | NIHR HTA funded trial – OpNonSTOP study; recruitment starts July 2024 |
| JLA no. 6 | Effectiveness of selective dorsal rhizotomy in cerebral palsy | NIHR doctoral fellowship started in 2024 |
| JLA no. 7 | Effectiveness of non-surgical treatment for children with cerebral palsy who can walk | NIHR HTA funded two commissioned trials, the SPELL study6 and the ROBUST study,7 both currently recruiting |
| JLA no. 8 and BSCOS elective no. 6 | Optimizing the screening for DDH | DDH research programme being developed with NIHR Academy Funding, with multiple doctorates; SMARTIE and SMARTER studies8 |
| BSCOS elective no. 1 | What is the optimal approach to treat severe stable SCFE - correction of deformity or pinning in situ? | NIHR HTA funded Recruiting Big BOSS Study9 BJJ editorial10 |
| BSCOS elective no. 2 | What is the optimal approach to treat Perthes’ disease - surgical containment, or nonoperative treatment? | NIHR doctoral fellowship and NIHR HTA funded Recruitment starts July 202411,12 |
| BSCOS elective no. 3 | Bone and joint infection; diagnosis and treatment | HTA/NIHR commissioned study on diagnostic imaging recruiting13,14 |
| BSCOS elective no. 7 | What are the approaches to the management of late presenting DDH in infants, and how may differences in treatment effect subsequent outcomes | DDH research programme being developed with NIHR Academy funding, SMARTIE and SMARTER studies |
| BSCOS elective no. 8 | Do infants with centred, but sonographically immature hips, benefit from neonatal splintage? | DDH research programme being developed with NIHR Academy funding, SMARTIE and SMARTER studies |
| BSCOS trauma no. 1 | Do children with medial epicondylar fractures benefit from surgical fixation compared to nonoperative treatment in terms of functional outcomes? | NIHR HTA funded, recruitment complete Results of SCIENCE study expected Spring 202515 Protocol16 Qualitative study published17 BJJ editorial18 |
| BSCOS trauma no. 3 | Do younger children with displaced distal radial fractures (either at the metaphysis or the physis) benefit from reduction with or without fixation, or do the results of nonoperative treatments offer similar functional and cosmetic outcomes | NIHR HTA funded Recruitment for CRAFFT Study reaching completion19 Qualitative study published20 BJJ editorial21 |
| BSCOS trauma no. 4 | Do children with minimally displaced fractures of the distal tibial physis benefit from operative treatment, compared to those treated with nonoperative treatment | NIHR HTA funded Recruitment for ODDSocks study starts Spring 202422 BJJ editorial23 |
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BJJ, The Bone & Joint Journal; BSCOS, British Society for Children’s Orthopaedic Surgery; DDH, developmental dysplasia of the hip; HTA, Health Technology Assessment; JLA, James Lind Alliance; NIHR, National Institute for Health and Care Research; SCFE, slipped capital femoral epiphysis.
Research in children’s orthopaedics is now flourishing, following the establishment of research priorities. This has engaged paediatric orthopaedic surgeons in the process of prioritization, allowing them to take ownership of this task and leading to the development of a research network, which is currently supporting multicentre trials and a growing community of doctoral students. Involving children and their families in the prioritization of research, with the support of the JLA, was key to getting funding.
While the priorities were intended to develop the landscape of research in this area in the UK, we are aware that they have been used to encourage funding for research throughout the world. For instance, randomized controlled trials concerning medial epicondylar (COMET)24 and distal radial fractures (DRIFT)25 have been funded by the USA National Institutes of Health. Although the UK priorities may broadly apply internationally, they may need to be tailored to the specific needs of different regions in order to reflect the needs of the patients and the funding bodies.
A clear research agenda has transformed children’s orthopaedic surgery in the UK, allowing it to become one of the subspecialties in which the most research is now undertaken, with considerable associated international involvement.
References
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Author contributions
T. Theologis: Writing – original draft.
D. C. Perry: Writing – review & editing.
Funding statement
The authors disclose receipt of the following financial or material support for the research, authorship, and/or publication of this article: D. C. Perry reports a NIHR Research Professorship, and funding from the NIHR Academy (NIHR301655).
ICMJE COI statement
D. C. Perry reports a NIHR Research Professorship, and funding from the NIHR Academy (NIHR301655).
Open access statement
This article is distributed under the terms of the Creative Commons Attributions (CC BY 4.0) licence (https://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium or format, provided the original author and source are credited.
This article was primary edited by J. Scott.
