CRediT contribution statement
Deb Pal (https://orcid.org/0000-0003-2655-0564): Conceptualisation (lead), Funding acquisition (lead), Project administration (lead), Methodology (equal), Supervision (lead), Writing – original draft (lead).
Paul Gringras (https://orcid.org/0000-0002-0495-3517): Conceptualisation (lead), Funding acquisition (equal), Methodology (equal), Supervision (equal), Writing – reviewing and editing (equal).
Aiswarya Anilkumar (https://orcid.org/0000-0001-5619-6207): Formal analysis (equal), Visualisation (lead), Writing – reviewing and editing (equal).
Lucy Bray (https://orcid.org/0000-0001-8414-3233): Conceptualisation (equal), Funding acquisition (equal), Investigation (equal), Methodology (equal), Resources (equal), Writing – reviewing and editing (equal).
Bernie Carter (https://orcid.org/0000-0001-5226-9878): Conceptualisation (equal), Funding acquisition (equal), Methodology (equal), Investigation (equal), Writing – reviewing and editing (equal).
Georgia Cook (https://orcid.org/0000-0002-1651-866X): Methodology (equal), Resources (equal), Investigation (equal), Formal analysis (equal), Visualisation (equal), Writing – reviewing and editing (equal).
Holly Crudgington (https://orcid.org/0000-0003-1048-4953): Investigation (equal), Formal analysis (equal), Visualisation (equal), Writing – reviewing and editing (equal).
Janet Currier (https://orcid.org/0009-0007-9928-2149): Conceptualisation (equal), Funding acquisition (equal), Writing – reviewing and editing (equal).
Will Hardy (https://orcid.org/0000-0001-5227-567X): Investigation (equal), Formal analysis (equal), Visualisation (equal), Writing – reviewing and editing (equal).
Dyfrig Hughes (https://orcid.org/0000-0001-8247-7459): Conceptualisation (equal), Funding acquisition (equal), Methodology (equal), Writing – reviewing and editing (equal).
Christopher Morris (https://orcid.org/0000-0002-9916-507X): Conceptualisation (equal), Funding acquisition (equal), Methodology (equal), Supervision (equal), Resources (equal), Investigation (equal), Formal analysis (equal), Writing – reviewing and editing (equal).
Deborah Roberts (https://orcid.org/0000-0002-3845-5786): Conceptualisation (equal), Funding acquisition (equal), Writing – reviewing and editing (equal).
Holly Saron (https://orcid.org/0000-0001-7563-3409): Investigation (equal), Formal analysis (equal), Writing – reviewing and editing (equal).
Catrin Tudur Smith (https://orcid.org/0000-0003-3051-1445): Conceptualisation (equal), Methodology (equal), Supervision (equal), Formal analysis (equal), Writing – reviewing and editing (equal).
Luci Wiggs (https://orcid.org/0000-0002-5697-6550): Conceptualisation (equal), Methodology (equal), Funding acquisition (equal), Resources (equal), Formal analysis (equal), Writing – reviewing and editing (equal).
Acknowledgements
We would like to sincerely thank all the participants and families who took part in the trial and other parts of the programme. We would also like to thank the CASTLE researchers and trial managers who contributed to study organisation: Lucy Stibbs-Eaton, Nadia al-Najjar, Rachael Martin, Amber Collingwood, Anna Smith, Agnès Tort, Eifiona Wood, Alison Rouncefield-Swales, Catherine Spowart, Anna Fiori, Sam Lyle, Tony Coffey, Morwenna Rogers, Kristina Dietz, Helen Hickey, Frances Gibbon, Fiona Tierney, Liam Whittle and Colin Dunkley. We are most grateful to Jo Conduit-Smith, Martyn Lewis, Desaline Joseph and Jeremy Parr for guidance from the Steering Committee and to Helen Cross, Alberto Verrotti, Izzy Coleman and the late Tony Johnson of the Independent Data and Safety Monitoring Committee.
Patient data statement
This work uses data provided by patients and collected by the NHS as part of their care and support. Using patient data is vital to improve health and care for everyone. There is huge potential to make better use of information from people’s patient records, to understand more about disease, develop new treatments, monitor safety, and plan NHS services. Patient data should be kept safe and secure, to protect everyone’s privacy, and it is important that there are safeguards to make sure that they are stored and used responsibly. Everyone should be able to find out about how patient data are used. #datasaveslives You can find out more about the background to this citation here: https://understandingpatientdata.org.uk/data-citation.
Data-sharing statement
All data requests should be submitted to the corresponding author for consideration. Access to anonymised data may be granted following review. Researchers requesting access should provide a clear justification for their intended use and agree to comply with all applicable data security and privacy policies.
Ethics statement
Work package 4 was originally approved as the CASTLE trial (IRAS ID 250324, 4 October 2019, REC 19/LO/0452, 23 September 2020). The CASTLE Sleep-E trial was then approved as a separate trial under WP4 (IRAS ID 289580, REC 21/EM/0205, 10 December 2021).
Information governance statement
King’s College London is committed to handling all personal information in line with the UK Data Protection Act (2018) and the General Data Protection Regulation (EU GDPR) 2016/679. Under the Data Protection legislation, King’s College London is the Data Controller, and you can find out more about how we handle personal data, including how to exercise your individual rights and the contact details for our Data Protection Officer here: @info-compliance@kcl.ac.uk. King’s College London ICO registration number: Z7915194.
Disclosure of interests
Full disclosure of interests: Completed ICMJE forms for all authors, including all related interests, are available in the toolkit on the NIHR Journals Library report publication page at https://doi.org/10.3310/GJDP1929.
Primary conflicts of interest: Deb Pal is chief investigator of the current research funded by the National Institute for Health Research (NIHR) (RP-PG-0615-20007). He is the coinventor of SleepSuite™, an iPad measure of sleep-related memory consolidation that was used as a secondary outcome measure in the CASTLE Sleep-E trial. Paul Gringras is cochief investigator of the current research funded by the National Institute for Health Research. He is the coinventor of SleepSuite™, an iPad measure of sleep-related memory consolidation that was used as a secondary outcome measure in the CASTLE Sleep-E trial. He has consulted for and received speaker fees from AGB, Pharmanord and Neurim in relation to the melatonin products they manufacture. Georgia Cook has received research funds from NIHR206382. Dyfrig Hughes has served on the NIHR HTA clinical evaluation and trials committee (31 December 2010–31 March 2016). Catrin Tudur Smith has served on the HTA commissioning committee (1 November 2015–30 September 2020).
Copyright and credit statement
Every effort has been made to obtain the necessary permissions for reproduction, to credit original sources appropriately and to respect copyright requirements. However, despite our diligence, we acknowledge the possibility of unintentional omissions or errors and we welcome notifications of any concerns regarding copyright or permissions.
Publications
Morris C, Dunkley C, Gibbon FM, Currier J, Roberts D, Rogers M, et al. Core Health Outcomes In Childhood Epilepsy (CHOICE): protocol for the selection of a core outcome set. Trials 2017;18:572. https://doi.org/10.1186/s13063-017-2323-7
Crudgington H, Rogers M, Bray L, Carter B, Currier J, Dunkley C, et al. Core Health Outcomes in Childhood Epilepsy (CHOICE): development of a core outcome set using systematic review methods and a Delphi survey consensus. Epilepsia 2019;60:857–71. https://doi.org/10.1111/epi.14735
Crudgington H, Rogers M, Morris H, Gringras P, Pal DK, Morris C. Epilepsy-specific patient-reported outcome measures of children’s health-related quality of life: a systematic review of measurement properties. Epilepsia 2020;61:230–48. https://doi.org/10.1111/epi.16430
Crudgington H, Collingwood A, Bray L, Lyle S, Martin R, Gringras P, et al. Mapping epilepsy-specific patient-reported outcome measures for children to a proposed core outcome set for childhood epilepsy. Epilepsy Behav 2020;112:107372. https://doi.org/10.1016/j.yebeh.2020.107372
Cook G, Gringras P, Hiscock H, Pal DK, Wiggs L. A qualitative investigation into what parents want from an online behavioural sleep intervention for children with epilepsy. Front Psychol 2021;12:628605. https://doi.org/10.3389/fpsyg.2021.628605
Wiggs L, Cook G, Hiscock H, Pal DK, Gringras P. Development and evaluation of the CASTLE trial online sleep intervention for parents of children with epilepsy. Front Psychol 2021;12:679804. https://doi.org/10.3389/fpsyg.2021.679804
Carter B, Cook G, Bray L, Collingwood A, Saron H, Rouncefield-Swales A. Parents’/caregivers’ fears and concerns about their child’s epilepsy: a scoping review. PLOS ONE 2022;17:e0274001. https://doi.org/10.1371/journal.pone.0274001
Al-Najjar N, Bray L, Carter B, Castle AP, Collingwood A, Cook G, et al. Changing Agendas on Sleep, Treatment and Learning in Epilepsy (CASTLE) Sleep-E: a protocol for a randomised controlled trial comparing an online behavioural sleep intervention with standard care in children with Rolandic epilepsy. BMJ Open 2023;13:e065769. https://doi.org/10.1136/bmjopen-2022-065769
Carter B, Bray L, Al-Najjar N, Piella AT, Tudur-Smith C, Spowart C, et al. The impact of parent treatment preference and other factors on recruitment: lessons learned from a paediatric epilepsy randomised controlled trial. Trials 2023;24:83. https://doi.org/10.1186/s13063-023-07091-9
Cook G, Bray L, Carter B, Gringras P, Morris C, Pal DK, et al. A cross-sectional survey of healthcare professionals supporting children and young people with epilepsy and their parents/carers: which topics are raised in clinical consultations and can healthcare professionals provide the support needed? Epilepsy Behav 2023;149:109543. https://doi.org/10.1016/j.yebeh.2023.109543
Cook G, Gringras P, Hiscock H, Pal DK, Wiggs L. ‘No one’s ever said anything about sleep’: a qualitative investigation into mothers’ experiences of sleep in children with epilepsy. Health Expect 2023;26:693–704. https://doi.org/10.1111/hex.13694
Conference abstracts
Crudgington H, Morris C, Dunkley C, Gibbon F, Currier J, Roberts D, et al. Core Health Outcomes in Childhood Epilepsy: Selection of a Core Outcome Set. British Paediatric Neurology Association Conference (BPNA), London, 2018.
Crudgington H, Rogers M, Bray L, Carter B, Currier J, Dunkley C, et al. Core Health Outcomes in Childhood Epilepsy (CHOICE) – Development of a Core Outcome Set. 13th European Congress on Epileptology, Vienna, 2018 poster. URL: www.professionalabstracts.com/ece2018/programmeece2018.pdf (accessed January 2025).
Crudgington H, Rogers M, Bray L, Carter B, Currier J, Dunkley C, et al. Core Health Outcomes In Childhood Epilepsy (CHOICE) Study: Development of a Core Outcome Set (COS) Using Systematic Review Methods and a Delphi Survey Consensus. Core Outcome Measures in Effectiveness Trials VI meeting, Amsterdam, 2018.
Crudgington H, Rogers M, Bray L, Carter B, Currier J, Dunkley C, et al. Core Health Outcomes In Childhood Epilepsy (CHOICE) Study: Development of a Core Outcome Set (COS) Using Systematic Review Methods and a Delphi Survey Consensus. European Academy for Childhood Disability (EACD) conference, Paris, 2019 poster. URL: https://edu.eacd.org/node/490 (accessed January 2025).
Crudgington H, Collingwood A, Bray L, Martin R, Lyle S, Gringras P, et al. Mapping Epilepsy-specific Patient-reported Outcome Measures for Children to a Core Outcome Set for Childhood Epilepsy. American Epilepsy Society 2020.
Crudgington H, Collingwood A, Rogers M, Morris H, Bray L, Carter B, et al. Core Health Outcomes in Childhood Epilepsy (CHOICE): Agreeing Critical Domains and Appraising How to Measure Them. International Alliance of Academies of Childhood Disability (IAACD) Better Together, online, 2021.
Crudgington H, Collingwood A, Rogers M, Morris H, Bray L, Carter B, et al. Developing a Core Outcome Set for Childhood Epilepsy. International Pediatric Sleep Association (IPSA), online, 2022.
Conference presentations
Cook G. Research Developments: Changing Agendas on Sleep Treatment and Learning in Childhood Epilepsy (CASTLE). Guy’s and St Thomas’ Foundation Trust: Autism 24/7: Domains and dimensions: towards trans-diagnostic interventions conference, online, 24–25 February 2022.
Cook G. Changing Agendas on Sleep Treatment and Learning in Childhood Epilepsy (CASTLE) Online Sleep. Intervention Development [Conference Presentation]. British Paediatric Sleep Society conference, online, 11 March 2022.
Cook G, Wiggs L. An Online Sleep Intervention for Children with Epilepsy as Part of the Creating Sleep Solutions: Translating Parents’ Concerns about Sleep and Their Child’s Seizures into Digital Therapeutics Symposium. International Pediatric Sleep Association, online, 27–29 October 2022.
Cook G, Bray L, Carter B, Gringras P, Morris C, Pal DK, et al. A Cross-Sectional Survey of Clinicians’ Experiences of Addressing the Topic of Sleep in Children and Young People with Epilepsy. British Sleep Society conference, Leeds, UK, 2–4 October 2023.
Conference posters
Wiggs L, Cook G, Hiscock H, Gringras P. Development of an Online Behavioural Sleep Intervention for Parents of Children with Epilepsy, for Use in the CASTLE Study Clinical Trial. British Sleep Society conference, Birmingham, UK, 21–23 November 2019.
Cook G, Gringras P, Pal DK, Wiggs L. ‘No One’s Ever Said Anything About Sleep’: A Qualitative Investigation into Parents’ Experiences of Sleep and Sleep Problem Management in Children with Epilepsy. Pediatric Sleep Medicine Conference, online, 1–3 December 2021.
Cook G, Gringras P, Hiscock H, Pal DK, Wiggs L. Parents’ Experiences of Sleep, Sleep Problems and Their Management in Children with Epilepsy. International Epilepsy Congress, Dublin, Ireland, 2–6 September 2023.
Cook G, Wiggs L, Hiscock H, Pal DK, Gringras P. Developing an Online Behaviourally Based Sleep Intervention for Parents of Children with Epilepsy for Use in a Large Multi-Site UK Based Randomised Control Trial. International Epilepsy Congress, Dublin, Ireland, 2–6 September 2023.
Cook G, Gringras P, Pal DK, Hiscock H, Wiggs L. Parental Cognitions about the Sleep of Children with Epilepsy and the Impact of a Behavioural Sleep Intervention on These Thoughts, Feelings, Beliefs and Attitudes. International Pediatric Sleep Association. Glasgow, Scotland, 26–28 April 2024.
Tierney F, Cook G, Gringras P, Pal DK, Wiggs L. Caregiver-Reported Sleep Characteristics of Children with Epilepsy and Their Caregivers in Independently Sleeping, Regular Cosleeping and Irregularly Cosleeping Families. International Pediatric Sleep Association. Glasgow, Scotland, 26–28 April 2024.
Wiggs L, Gringras P, Pal DK, Hiscock H, Cook G. The CASTLE Online Sleep Intervention (COSI) for Children with Epilepsy: Parents’ Use of COSI in a Clinical Trial. International Pediatric Sleep Association. Glasgow, Scotland, 26–28 April 2024.
Cook G. The Coproduction of a Behaviourally Based Online Sleep Intervention for Parents of Children with Epilepsy for Use in a Multisite Randomised Control Trial in the UK. Epilepsy Research Institute: Emerging Leaders Symposium, London, UK, 21 May 2024.
Blogs and videos
https://castlestudy.org.uk/news/
Epilepsy Sparks podcast: www.torierobinson.com/epilepsy-sparks-insights/georgia-cook-communication-barriers-in-epilepsy-research
Epilepsy Sparks podcast: www.torierobinson.com/epilepsy-sparks-insights/georgia-cook-understanding-researchers-patients-people-with-epilepsy
Epilepsy Sparks interview at CASTLE conference in Manchester, July 2024: www.youtube.com/watch?v=QL4VSNtpglM
Epilepsy Sparks podcast: www.torierobinson.com/epilepsy-sparks-insights/deb-pal-paul-gringras-exploring-the-links-between-sleep-epilepsy-castle
When Epilepsy Walks with You: www.youtube.com/watch?v=Em-IAQjnEH8
Community engagement
https://castlestudy.org.uk/events/castle-winter-family-engagement-day/
https://castlestudy.org.uk/events/castle-advisory-panel-family-day-at-knowsley-safari-park/
Disclaimers
This article presents independent research funded by the National Institute for Health and Care Research (NIHR). The views and opinions expressed by authors in this publication are those of the authors and do not necessarily reflect those of the NHS, the NIHR, CCF, PGfAR or the Department of Health and Social Care. If there are verbatim quotations included in this publication the views and opinions expressed by the interviewees are those of the interviewees and do not necessarily reflect those of the authors, those of the NHS, the NIHR, the PGfAR programme or the Department of Health and Social Care.