UK research priority setting for childhood neurological conditions

Jill Cadwgan; Jane Goodwin; Barbara Babcock; Molly Brick; Richard Chin; Ava Easton; Ben Green; Siobhan Hannan; Rhys P D Inward; Suzannah Kinsella; Callum King; Manju A Kurian; Philip Levine; Andrew Mallick; Jeremy Parr; Carol Anne Partridge; Sam Amin; Dan Lumsden; J Helen Cross; Ming J Lim; UK Childhood Neurological Disorders PSP Group

doi:10.1111/dmcn.16021

UK research priority setting for childhood neurological conditions

Dev Med Child Neurol. 2024 Dec;66(12):1590-1599. doi: 10.1111/dmcn.16021. Epub 2024 Jul 16.

Authors

Jill Cadwgan¹, Jane Goodwin², Barbara Babcock³, Molly Brick⁴, Richard Chin^{5

6}, Ava Easton⁷, Ben Green⁴, Siobhan Hannan⁴, Rhys P D Inward⁸, Suzannah Kinsella⁹, Callum King⁴, Manju A Kurian^{10

11}, Philip Levine⁴, Andrew Mallick¹², Jeremy Parr², Carol Anne Partridge¹³, Sam Amin¹⁴, Dan Lumsden^{1

15}, J Helen Cross^{16

17}, Ming J Lim^{1

18}; UK Childhood Neurological Disorders PSP Group⁴

Collaborators

UK Childhood Neurological Disorders PSP Group:
Anne-Marie Childs, Vijeya Ganesan, Anthony Hart, Rachel Kneen, Robert McFarland, Evangeline Wassmer, William P Whitehouse, Ruth Williams, Tom Arichi, Vicky Burford, Sarah Crombie, Laavanya Damodaran, Paul Eunson, Paul Gringras, Anne Gordon, Emily Harrop, Anthony Hart, Catherine Martin, Carla Rush, Catherine Tuffrey

Affiliations

¹ Children's Neurosciences, Evelina London Children's Hospital, Guy's and St Thomas' NHS Foundation Trust, London, UK.
² Population Health Sciences Institute, Newcastle University, Newcastle upon Tyne, UK.
³ Transverse Myelitis Society, Brentford, UK.
⁴ British Paediatric Neurology Association, London, UK.
⁵ Centre for Clinical Brain Sciences, University of Edinburgh, Edinburgh, UK.
⁶ Royal Hospital for Children and Young People, Edinburgh, UK.
⁷ Department of Clinical Infection, Microbiology & Immunology, University of Liverpool, Liverpool, UK.
⁸ Department of Biology, University of Oxford, Oxford, UK.
⁹ The James Lind Alliance, National Institute for Health and Care Research, School of Healthcare Enterprise and Innovation, University of Southampton, Southampton, UK.
¹⁰ Developmental Neurosciences, Zayed Centre for Research into Rare Disease in Children, GOS-Institute of Child Health, University College London, London, UK.
¹¹ Department of Neurology, Great Ormond Street Hospital, London, UK.
¹² Department of Paediatric Neurology, Bristol Royal Hospital for Children, Bristol, UK.
¹³ CDKL5 UK, UK.
¹⁴ University Hospitals Bristol and Weston NHS Foundation Trust, Bristol, UK.
¹⁵ Early Life Imaging, School of Biomedical Engineering and Imaging Sciences, Faculty of Life Sciences and Medicine, King's College London, London, UK.
¹⁶ Developmental Neurosciences Research and Teaching Department, University College London NIHR BRC Great Ormond Street Institute of Child Health, London, UK.
¹⁷ Paediatric Neurosciences, Great Ormond Street Hospital for Children, London, UK.
¹⁸ Women and Children's Health, School of Life Course & Population Sciences, Faculty of Life Sciences and Medicine, King's College London, London, UK.

PMID: 39014885
DOI: 10.1111/dmcn.16021

Abstract

Aim: To identify research priorities regarding the effectiveness of interventions for children and young people (CYP) with childhood neurological conditions (CNCs). These include common conditions such as epilepsies and cerebral palsy, as well as many rare conditions.

Method: The National Institute for Health and Care Research (NIHR) and the James Lind Alliance (JLA) champion and facilitate priority setting partnerships (PSPs) between patients, caregivers, and clinicians (stakeholders) to identify the most important unanswered questions for research (uncertainties). A NIHR-JLA and British Paediatric Neurology Association collaboration used the JLA PSP methodology. This consisted of two surveys to stakeholders: survey 1 (to identify uncertainties) and survey 2 (a prioritization survey). The final top 10 priorities were agreed by consensus in a stakeholder workshop.

Results: One hundred and thirty-two charities and partner organizations were invited to participate. In survey 1, 701 participants (70% non-clinicians, including CYP and parent and caregivers) submitted 1800 uncertainties from which 44 uncertainties were identified for prioritization in survey 2; from these, 1451 participants (83% non-clinicians) selected their top 10 priorities. An unweighted amalgamated score across participant roles was used to select 26. In the final workshop, 14 health care professionals, 11 parent and caregivers, and two CYP ranked the 26 questions to finalize the top 10 priorities. Ten top priority questions were identified regarding interventions to treat CYP with CNCs and their associated comorbidities, for example, sleep, emotional well-being, and distressing symptoms.

Interpretation: The results of this study will inform research into the effectiveness of interventions for children with neurological conditions.

MeSH terms

Adolescent
Biomedical Research
Child
Female
Health Priorities
Humans
Male
Nervous System Diseases* / therapy
Research
Surveys and Questionnaires
United Kingdom