Outcome domains and outcome measures used in studies assessing the effectiveness of interventions to manage non-respiratory sleep disturbances in children with neurodisabilities: a systematic review

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Objectives: To assess whether a core outcome set is required for studies evaluating the effectiveness of interventions for non-respiratory sleep disturbances in children with neurodisabilities. Design: Survey of outcome measures used in primary studies identified by a systematic review. Data sources: ASSIA; CENTRAL; Cochrane Database of Systematic Reviews; Conference Proceedings Citation Index; CINAHL; DARE; Embase; HMIC; MEDLINE; MEDLINE In-Process; PsycINFO; Science Citation Index; Social Care Online; Social Policy & Practice; ClinicalTrials.gov; WHO International Clinical Trials Registry Platform (ICTRP); and the UK Clinical Trials Gateway were searched up to February 2017. Eligibility criteria: Studies evaluating pharmacological or non-pharmacological interventions for children (≤ 18 years old) with a neurodisability and experiencing non-respiratory sleep disturbance. Data extraction and synthesis: Outcomes related to child and parent sleep-related outcomes; measures of perceived parenting confidence, efficacy or understanding of sleep management; child-related quality of life, daytime behaviour and cognition; parent/carer outcomes; and adverse events were listed from each study and categorised into domains. Results: Thirty-nine studies (13 melatonin and 26 non-pharmacological) assessed five core outcome areas: child sleep, other child outcomes, parent outcomes, adverse events and process measures. There were 54 different measures of child related sleep across five domains: global measures; sleep initiation; maintenance; scheduling; and other outcomes. The most commonly reported measure in melatonin studies was total sleep time (n=12; 92%); and for non-pharmacological studies was the parent-reported Child Sleep Habits Questionnaire (CSHQ; 58%), both classified as global measures. Fifteen non-pharmacological (58%) and four pharmacological studies (31%) reported child outcomes other than sleep. The domains assessed (using 29 different measures) were child behaviour, quality of life, ADHD symptoms, cognition, school-related, and other. One pharmacological and 14 non-pharmacological (54%) studies reported parent outcomes (17 different measures). Eleven melatonin studies (85%) recorded adverse events, with variation in how data were collected and reported. One non-pharmacological study reported an explicit method of collecting on adverse events. Several process measures were reported, related to adherence, feasibility of delivery, acceptability and experiences of receiving the intervention. Conclusions: There is a lack of consistency between studies in the outcome measures used to assess the effectiveness of interventions for non-respiratory sleep disturbances in children with neurodisabilities. A minimum core outcome set, with international consensus, should be developed in consultation with parents, children and young people, and those involved in supporting families. Registration number systematic review PROSPERO (CRD42016034067)
Original languageEnglish
Article numbere027205
Number of pages12
JournalBMJ Open
Publication statusPublished - 19 Jun 2019

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