STUDY OBJECTIVES:The objectives of the study were (1) to compare both actigraphy and questionnaire-assessed sleep quality and timing in children with Smith-Magenis syndrome (SMS) to a chronologically age-matched typically developing (TD) group and (2) to explore associations between age, nocturnal and diurnal sleep quality, and daytime behavior.

METHODS:Seven nights of actigraphy data were collected from 20 children with SMS (mean age 8.70; SD 2.70) and 20 TD children. Daily parent/teacher ratings of behavior and sleepiness were obtained. Mixed linear modeling was used to explore associations between total sleep time and daytime naps and behavior.

RESULTS:Sleep in children with SMS was characterized by shorter total sleep time (TST), extended night waking, shorter sleep onset, more daytime naps, and earlier morning waking compared to the TD group. Considerable inter-daily and inter-individual variability in sleep quality was found in the SMS group, so caution in generalizing results is required. An expected inverse association between age and TST was found in the TD group, but no significant association was found for the SMS group. No between-group differences in sleep hygiene practices were identified. A bidirectional negative association between TST and nap duration was found for the SMS group. In the SMS group, increased afternoon sleepiness was associated with increased irritability (p = .007) and overactivity (p = .005).

CONCLUSION:These findings evidence poor sleep quality in SMS and the need to implement evidence-based interventions in this population.

译文

学习目标 : 该研究的目的是 (1) 将史密斯-马格尼斯综合征 (SMS) 儿童的活动记录和问卷评估的睡眠质量和时间与年龄匹配的典型发育 (TD) 组进行比较; (2) 探索年龄,夜间和昼夜睡眠质量之间的关联,和白天的行为。
方法 : 从20名SMS儿童 (平均年龄8.70岁; SD 2.70岁) 和20名TD儿童中收集了七个晚上的活动记录数据。获得了父母/老师对行为和嗜睡的每日评分。混合线性模型用于探索总睡眠时间与白天小睡和行为之间的关联。
结果 : SMS儿童的睡眠特征是总睡眠时间 (TST) 较短,夜间清醒时间延长,睡眠发作时间缩短,与TD组相比,白天小睡更多,早上醒来更多。在SMS组中发现睡眠质量存在相当大的日常和个体间差异,因此在概括结果时需要谨慎。在TD组中发现了年龄与TST之间的预期反向关联,但在SMS组中未发现显着关联。未发现睡眠卫生习惯的组间差异。对于SMS组,发现TST和nap持续时间之间存在双向负相关。在SMS组中,下午嗜睡增加与烦躁 (p = .007) 和过度活跃 (p = .005) 有关。
结论 : 这些发现表明SMS的睡眠质量较差,并且需要在该人群中实施循证干预措施。

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