Children and young people’s contributions to public involvement and engagement activities in health-related research: A scoping review

Background: There has been an increasing interest in how children and young people can be involved in patient and public involvement and engagement (PPIE) in health research. However, relatively little robust evidence exists about which children and young people are reported as being involved or excluded from PPIE; the methods reported as being used to involve them in PPIE; and the reasons presented for their involvement in PPIE and what happens as a result. We performed a scoping review to identify, synthesise and present what is known from the literature about patient and public involvement and engagement activities with children and young people in health related research. Methods: Relevant studies were identified by searches in Scopus, Medline, CINAHL, Cochrane and PsychInfo databases, and hand checking of reference lists and grey literature. An adapted version of the Guidance for Reporting Involvement of Patients and the Public (GRIPP2) was used as a framework to collate the data. Two reviewers independently screened articles and decisions were consensually made. Main findings: A total of 9805 references were identified (after duplicates were removed) through the literature search, of which 233 full-text articles were assessed for eligibility. Forty studies published between 2000 and 2019 were included in the review. The review reveals ambiguities in the quality of reporting of PPIE with children with clear reporting on demographics and health conditions. The review found that children and young people were commonly involved in multiple stages of research but there was also significant variation in the level at which children and young people were involved in PPIE. Evaluation of the impact of children and young people’s involvement in PPIE was limited. Conclusions: Consultation, engagement and participation can all offer children and young people worthwhile ways of contributing to research with the level, purpose and impact of involvement determined by the children and young people themselves. However, careful decisions need to be made to ensure that it is suited to the context, setting and focus so that the desired PPIE impacts are achieved. Improvements should be made to the evaluation and reporting of PPIE in research. This will help researchers and funders to better understand the benefits, challenges and impact of PPIE with children and young people on health research

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

This is the final version. Available from BMJ Publishing Group via the DOI in this record. INTRODUCTION: Sleep and epilepsy have an established bidirectional relationship yet only one randomised controlled clinical trial has assessed the effectiveness of behavioural sleep interventions for children with epilepsy. The intervention was successful, but was delivered via face-to-face educational sessions with parents, which are costly and non-scalable to population level. The Changing Agendas on Sleep, Treatment and Learning in Epilepsy (CASTLE) Sleep-E trial addresses this problem by comparing clinical and cost-effectiveness in children with Rolandic epilepsy between standard care (SC) and SC augmented with a novel, tailored parent-led CASTLE Online Sleep Intervention (COSI) that incorporates evidence-based behavioural components. METHODS AND ANALYSES: CASTLE Sleep-E is a UK-based, multicentre, open-label, active concurrent control, randomised, parallel-group, pragmatic superiority trial. A total of 110 children with Rolandic epilepsy will be recruited in outpatient clinics and allocated 1:1 to SC or SC augmented with COSI (SC+COSI). Primary clinical outcome is parent-reported sleep problem score (Children's Sleep Habits Questionnaire). Primary health economic outcome is the incremental cost-effectiveness ratio (National Health Service and Personal Social Services perspective, Child Health Utility 9D Instrument). Parents and children (≥7 years) can opt into qualitative interviews and activities to share their experiences and perceptions of trial participation and managing sleep with Rolandic epilepsy. ETHICS AND DISSEMINATION: The CASTLE Sleep-E protocol was approved by the Health Research Authority East Midlands (HRA)-Nottingham 1 Research Ethics Committee (reference: 21/EM/0205). Trial results will be disseminated to scientific audiences, families, professional groups, managers, commissioners and policymakers. Pseudo-anonymised individual patient data will be made available after dissemination on reasonable request. TRIAL REGISTRATION NUMBER: ISRCTN13202325.National Institute for Health and Care Research (NIHR)National Health and Medical Research Council (NHMRC, Australia)Victorian Government’s Operational Infrastructure Support Progra

The impact of parent treatment preference and other factors on recruitment: lessons learned from a paediatric epilepsy randomised controlled trial

This is the final version. Available on open access from BMC via the DOI in this recordAvailability of data and materials: The CASTLE trial was terminated after the pilot. The datasets used and/or analysed during the CASTLE pilot as well as select trial materials are available from the corresponding author (DP) on reasonable request (see also pages 53-54 of the attached protocol). The information from the consultation with the health professionals is not available as this is not research data.Background In paediatric epilepsy, the evidence of effectiveness of antiseizure treatment is inconclusive for some types of epilepsy. As with other paediatric clinical trials, researchers undertaking paediatric epilepsy clinical trials face a range of challenges that may compromise external validity Main body In this paper, we critically reflect upon the factors which impacted recruitment to the pilot phase of a phase IV unblinded, randomised controlled 3×2 factorial trial examining the effectiveness of two antiseizure medications (ASMs) and a sleep behaviour intervention in children with Rolandic epilepsy. We consider the processes established to support recruitment, public and patient involvement and engagement (PPIE), site induction, our oversight of recruitment targets and figures, and the actions we took to help us understand why we failed to recruit sufficient children to continue to the substantive trial phase. The key lessons learned were about parent preference, children’s involvement and collaboration in decision-making, potential and alternative trial designs, and elicitation of stated preferences pre-trial design. Despite pre-funding PPIE during the trial design phase, we failed to anticipate the scale of parental treatment preference for or against antiseizure medication (ASMs) and consequent unwillingness to be randomised. Future studies should ensure more detailed and in-depth consultation to ascertain parent and/or patient preferences. More intense engagement with parents and children exploring their ideas about treatment preferences could, perhaps, have helped predict some recruitment issues. Infrequent seizures or screening children close to natural remission were possible explanations for non-consent. It is possible some clinicians were unintentionally unable to convey clinical equipoise influencing parental decision against participation. We wanted children to be involved in decisions about trial participation. However, despite having tailored written and video information to explain the trial to children we do not know whether these materials were viewed in each consent conversation or how much input children had towards parents’ decisions to participate. Novel methods such as parent/patient preference trials and/or discrete choice experiments may be the way forward. Conclusion The importance of diligent consultation, the consideration of novel methods such as parent/patient preference trials and/or discrete choice experiments in studies examining the effectiveness of ASMs versus no-ASMs cannot be overemphasised even in the presence of widespread clinician equipoise.National Institute for Health Research (NIHR

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

This is the final version. Available from BMJ Publishing Group via the DOI in this record. INTRODUCTION: Sleep and epilepsy have an established bidirectional relationship yet only one randomised controlled clinical trial has assessed the effectiveness of behavioural sleep interventions for children with epilepsy. The intervention was successful, but was delivered via face-to-face educational sessions with parents, which are costly and non-scalable to population level. The Changing Agendas on Sleep, Treatment and Learning in Epilepsy (CASTLE) Sleep-E trial addresses this problem by comparing clinical and cost-effectiveness in children with Rolandic epilepsy between standard care (SC) and SC augmented with a novel, tailored parent-led CASTLE Online Sleep Intervention (COSI) that incorporates evidence-based behavioural components. METHODS AND ANALYSES: CASTLE Sleep-E is a UK-based, multicentre, open-label, active concurrent control, randomised, parallel-group, pragmatic superiority trial. A total of 110 children with Rolandic epilepsy will be recruited in outpatient clinics and allocated 1:1 to SC or SC augmented with COSI (SC+COSI). Primary clinical outcome is parent-reported sleep problem score (Children's Sleep Habits Questionnaire). Primary health economic outcome is the incremental cost-effectiveness ratio (National Health Service and Personal Social Services perspective, Child Health Utility 9D Instrument). Parents and children (≥7 years) can opt into qualitative interviews and activities to share their experiences and perceptions of trial participation and managing sleep with Rolandic epilepsy. ETHICS AND DISSEMINATION: The CASTLE Sleep-E protocol was approved by the Health Research Authority East Midlands (HRA)-Nottingham 1 Research Ethics Committee (reference: 21/EM/0205). Trial results will be disseminated to scientific audiences, families, professional groups, managers, commissioners and policymakers. Pseudo-anonymised individual patient data will be made available after dissemination on reasonable request. TRIAL REGISTRATION NUMBER: ISRCTN13202325.National Institute for Health and Care Research (NIHR)National Health and Medical Research Council (NHMRC, Australia)Victorian Government’s Operational Infrastructure Support Progra