Gwilliam Kate, Joyce Anna, Dimitriou Dagmara
Lifespan Learning and Sleep Laboratory, UCL-Institute of Education, London, UK.
School of Psychotherapy & Psychology, Regent's University London, London, UK.
Res Dev Disabil. 2020 Sep;104:103658. doi: 10.1016/j.ridd.2020.103658. Epub 2020 Jun 3.
Children with neurodevelopmental disorders commonly experience sleep problems. Williams Syndrome (WS), a rare genetic disorder characterised by a complex, uneven cognitive profile, is no exception. Compared with children with typical development (TD), school-aged children with WS experience significant sleep disruption: shorter sleep duration, more night wakings, greater bedtime resistance and excessive daytime tiredness. In children with TD, sleep problems impede optimal daytime functioning. In WS, this could compound existing difficulties. Few studies have examined sleep in very young children with WS and little is known about the early emergence of sleep problems in this population. To date, studies have been based on parent-report and no studies have objectively assessed sleep patterns using longitudinal approach in toddlers with WS. Thus, the current study sought to objectively explore sleep patterns in toddlers with WS. Parents of 38 children (13 WS, 25 TD) completed the Brief Infant Screening Questionnaire and the Medical and Demographics Questionnaire and sleep patterns were assessed using actigraphy. Data were collected longitudinally at ages 18, 24 and 30 months. Significant sleep disturbances were present in WS from 18 months old. Sleep duration, as measured by actigraphy, was significantly shorter in WS at all ages and, furthermore, parents of children with WS reported more night wakings, longer settling times and high levels of parental involvement. Crucially, whereas actigraphy showed developmental improvements in sleep quality in TD, no longitudinal changes were found in WS. Findings could be instrumental in working towards instigating appropriate, timely sleep management in this group, thus improving outcomes for children and their families.
患有神经发育障碍的儿童通常会出现睡眠问题。威廉姆斯综合征(WS)是一种罕见的遗传性疾病,其特征是认知特征复杂且不均衡,也不例外。与发育正常(TD)的儿童相比,患有WS的学龄儿童存在明显的睡眠障碍:睡眠时间缩短、夜间醒来次数增多、就寝时间抗拒性增强以及白天过度疲劳。在TD儿童中,睡眠问题会妨碍白天的最佳功能。在WS中,这可能会使现有的困难更加复杂。很少有研究调查过非常年幼的WS儿童的睡眠情况,对于这一人群中睡眠问题的早期出现知之甚少。迄今为止,研究都是基于家长报告,没有研究使用纵向研究方法客观评估WS幼儿的睡眠模式。因此,本研究旨在客观探索WS幼儿的睡眠模式。38名儿童(13名WS儿童,25名TD儿童)的家长完成了简短婴儿筛查问卷和医疗与人口统计学问卷,并使用活动记录仪评估睡眠模式。在18、24和30个月大时纵向收集数据。WS儿童从18个月大时就出现了明显的睡眠障碍。通过活动记录仪测量,WS儿童在所有年龄段的睡眠时间都明显较短,此外,WS儿童的家长报告夜间醒来次数更多、入睡时间更长以及家长参与程度较高。至关重要的是,虽然活动记录仪显示TD儿童的睡眠质量有发育改善,但在WS儿童中未发现纵向变化。这些发现可能有助于为该群体制定适当、及时的睡眠管理措施,从而改善儿童及其家庭的状况。