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

Abstract

ObjectivesTo assess whether a core outcome set is required for studies evaluating the effectiveness of interventions for non-respiratory sleep disturbances in children with neurodisabilities.DesignSurvey of outcome measures used in primary studies identified by a systematic review.Data sourcesASSIA, 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 and the UK Clinical Trials Gateway were searched up to February 2017.Eligibility criteriaStudies evaluating pharmacological or non-pharmacological interventions for children (≤18 years old) with a neurodisability and experiencing non-respiratory sleep disturbance.Data extraction and synthesisOutcome measures were listed from each study and categorised into domains.ResultsThirty-nine studies assessed five core outcome areas: child sleep, other child outcomes, parent outcomes, adverse events and process measures. There were 54 different measures of child sleep across five domains: global measures; sleep initiation; maintenance; scheduling; and other outcomes. Fifteen non-pharmacological (58%) and four pharmacological studies (31%) reported child outcomes other than sleep using 29 different measures.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.ConclusionsThere 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.PROSPERO registration numberCRD42016034067

Youth and family engagement in childhood disability evidence syntheses: A scoping review

“…The outcomes of patient engagement reported in articles included in this review are consistent with that of Cottrell et al (2014), which support the benefits of youth and family engagement including improved understanding of the review question context and increased applicability of the results (Gonzalez et al, 2020;McConachie et al, 2015;McDaid et al, 2019). Authorship on publications and feelings of empowerment were also reported in the evidence syntheses included in this review (Camden et al, 2015;Gonzalez et al, 2020;Willis et al, 2017), and this finding is consistent with primary studies demonstrating empowerment as a key outcome of stakeholder engagement (Black et al, 2018;Ray & Miller, 2017).…”

“…Five articles reported recruitment of youth and family onto the evidence synthesis team through an established network or an organization that typically had a specific mandate for youth and family engagement. These included Peninsula Cerebra Research Unit ( n = 3) (Bailey et al, 2015; Blake et al, 2015; McConachie et al, 2015) and CHILD‐BRIGHT ( n = 1) (Gonzalez et al, 2020); and one paper indicated that family was ‘recruited from an existing parent consultation group’ (McDaid et al, 2019, p. 3). Additional details about how youth and families were recruited were not reported in these articles.…”

“…The engagement of youth and/or family at specific stages within the evidence synthesis projects is reported below. Furthermore, there was one study in which youth and family were reported to engage in all stages of the project (Gonzalez et al, 2020); a second study where stakeholders were invited to participate in three review team meetings, the timing and nature of which was unclear (McDaid et al, 2019); and a third study in which engagement occurred prior to the project to develop the MeASURe conceptual framework for evaluating outcomes of ASD that could be applied during the review (McConachie et al, 2015). In two reports, a parent was included as a member of the team and included as co‐authors on the resulting papers (Camden et al, 2015; Gonzalez et al, 2020).…”

“…Almost all articles (n = 8) reported engagement in the analysis and reporting stages with youth and/or family involved in the interpretation of results through their own experiential lens (Bailey et al, 2015;Blake et al, 2015;Camden et al, 2015;Forsythe et al, 2014;Gonzalez et al, 2020;Krieger et al, 2018;McDaid et al, 2019;Willis et al, 2017).…”

Symptom network analysis of the sleep disorders diagnostic criteria based on the clinical text of the ICSD‐3

“…An objective is to propose a methodology to measure and report COS and to encourage the collection of large amounts of data in order to improve their generalisability and credibility, and especially to encourage the widespread participation of international stakeholders (Chevance et al, 2020). Further research might sustain the development of COS for Sleep Medicine (McDaid et al, 2019; Wanyan et al, 2020).…”