ArticleOrphanet journal of rare diseases2022
Caregivers' experience of sleep management in Smith-Magenis syndrome: a mixed-methods study.
Article in Orphanet journal of rare diseases, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers, 1 of them a synthesis that pooled it.
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Who cites it
7 citing papers in PubMed, 1 synthesis or guideline pooled it, 15 citations in OpenAlex.
- Development and content evaluation of a multidimensional coping model-based health education manual for dementia care support.Frontiers in public health · 2026Pooled it
- The interactions between sleep difficulties and clinical features in children with rare genetic syndromes: a structural equation modelling approach.BMC pediatrics · 2026Article
- Sleep disorders in children/adolescents with neurodevelopmental and neurological disorders: what evidences do we have with the use of non-pharmacological interventions?Frontiers in sleep · 2026Review
- Subjective sleep assessment in individuals withJournal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine · 2024Article
- Exploring an objective measure of overactivity in children with rare genetic syndromes.Journal of neurodevelopmental disorders · 2024Article
- Blind to the perils of pursuing food: Behaviors of individuals with Smith-Magenis Syndrome.Genetics in medicine open · 2024Article
- The developmental trajectory of sleep in children with Smith-Magenis syndrome compared to typically developing peers: a 3-year follow-up study.Sleep advances : a journal of the Sleep Research Society · 2023Article
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Authors and funding
7 authors at 5 institutions in 2 countries.
Funding
Abstract
backgroundSmith-Magenis syndrome (SMS) is a rare genetic syndrome associated with a unique profile of early morning waking and daytime sleepiness. Children with SMS evidence high rates of self-injury and aggression and have a preference for adult over peer attention, with strong motivation to interact with a particular caregiver. In addition, people with SMS have lower adaptive functioning skills relative to cognitive abilities and demonstrate high levels of impulsivity. Taken together, these factors may result in individuals being awake overnight requiring vigilant caregiver supervision. Despite these complexities, no study has described the strategies caregivers take to keep their children with SMS safe overnight or considered the impact of these experiences on caregivers or the wider family.
methodsThe current study used a mixed-methods approach to consider sleep management strategies and challenges for caregivers of people with SMS at different ages. Caregivers completed an international online survey about sleep management and related difficulties, use of interventions and access to services and support. Semi-structured interviews were conducted with 14 caregivers in the UK to increase understanding of caregiver experiences and priorities for change in the UK context. Interviews were transcribed verbatim and coded using thematic analysis.
resultsEvidence from the online survey (n = 40) revealed wide-ranging impacts of poor sleep on the person with SMS and the wider family. Only 5% of caregivers reported that the sleep problems had no impact on their child, and 76% reported a moderately or extremely significant impact on themselves. For some individual caregivers, sleep management difficulties improved over time whereas for others no change was reported. Weekly respite emerged as the ideal provision for 49% of caregivers, although only 14% had access to this. The majority of caregivers (54%) received no respite. Thematic analysis of qualitative interviews revealed interactions between aspects of the behavioural phenotype of SMS which may contribute to complex and unusual presentations in relation to sleep management and safety.
conclusionsCaregivers' priorities for sleep management and support were delineated, with key implications for services in terms of the use of SMS-sensitive strategies and respite provision.
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