Public involvement and co-design of longitudinal studies of sleep health alongside young people with rare genetic conditions.

backgroundThe mechanisms underpinning associations between sleep and psychiatric conditions are poorly understood, partly due to challenges with longitudinal sleep studies outside the laboratory. Children and young people with rare genetic conditions caused by micro-deletions or -duplications (Copy Number Variants or CNVs) have increased risk of disrupted sleep and poorer neurodevelopmental (ND) outcomes. The 'Sleep Detectives' study aims to investigate this by tracking behavioural and neurophysiological signatures of sleep health in young people with ND risk or ND-CNVs. To optimally achieve this, we worked with families with ND-CNVs and charity partners to co-design our tools, methods, study protocol, and materials.

methodWe established a Lived Experience Advisory Group (LEAP): nine parents, 13 children and young people with ND-CNVs, and representatives of UK charities Max Appeal and Unique. Together, the research team and LEAP co-designed two in-person family workshops to collect feedback on acceptability of sleep monitoring devices, the design of bespoke cognitive tasks, and overall study protocol. Informal interviews and surveys enabled LEAP members and researchers to reflect and learn from their Patient/Public Involvement (PPI) experiences.

resultsKey outputs included pre-workshop information materials, and multiple insights and recommendations, all of which were incorporated iteratively in refining 16 different aspects of the main study design. These included more flexibility in data collection, selection of sleep devices, customisation of cognitive tasks, and improved document language. In a survey, 100% of workshop respondents (15/15) were positive or very positive about the overall study. The PPI process was highly valued by LEAP members, workshop attendees, and the research team. One investigator described it as "reinvigorating my love of research by helping me focus on science that matters". Participating families also established peer support networks.

conclusionsInvolving families affected by ND-CNVs in study co-design maximised opportunities for acceptability, accessibility and scalability. The researchers gained inspiration and deeper understanding of the impact of ND-CNVs on families. Families gained awareness about research, established connections with each other and peer support, and were enthusiastic about future research involvement. This experience empowered families to engage more deeply with the research process and made the PPI work more impactful and inclusive.