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雷特综合征中的睡眠障碍:影响与管理,包括睡眠卫生措施的应用。

Sleep disturbances in Rett syndrome: Impact and management including use of sleep hygiene practices.

作者信息

Boban Sharolin, Leonard Helen, Wong Kingsley, Wilson Andrew, Downs Jenny

机构信息

Telethon Kids Institute, The University of Western Australia, West Perth, Australia.

Respiratory Medicine, Princess Margaret Hospital for Children, Perth, Australia.

出版信息

Am J Med Genet A. 2018 Jul;176(7):1569-1577. doi: 10.1002/ajmg.a.38829. Epub 2018 Apr 28.

Abstract

Sleep disturbances are debilitating for individuals with Rett syndrome (RTT) and their families yet the evidence base for management is poor. We investigated management strategies and their relationships with sleep problems. Data were provided by 364/461 (79%) families with a child with RTT and registered with the International RTT Phenotype Database. Logistic regression models were used to investigate relationships between impacts of sleep problems on the child and family with age group, mutation type, medication type, and sleep hygiene score. Linear regression models were used to estimate the association of disorders of initiating and maintaining sleep (DIMS) with age group, mutation type, medication type, and sleep hygiene. Among those who ever had difficulty falling asleep or night waking, use of any medication was associated with higher odds of moderate/major impact sleep problems (relative to minor/no impact) for the affected child and the family, as well as higher DIMS scores, when compared with the no treatment/nonmedication group accounting for the effects of age, mutation type, and sleep hygiene score. Better use of sleep hygiene practices was associated with lower odds of moderate/major impact on the family (odds ratio 0.60, 95% confidence intervals [CIs] 0.37, 0.98) and lower DIMS scores (geometric mean ratio 0.86, 95%CI 0.80, 0.92) compared with poorer use after adjusting for covariates. Attention to sleep hygiene remains an important management strategy for sleep problems in RTT. Further prospective research is required to investigate efficacy of pharmaceutical treatments.

摘要

睡眠障碍对患有雷特综合征(RTT)的个体及其家庭来说是极为不利的,但针对该病症的管理证据基础却很薄弱。我们对管理策略及其与睡眠问题的关系进行了调查。数据由461个家庭中的364个(79%)提供,这些家庭育有一名患有RTT的儿童,并已在国际RTT表型数据库中注册。采用逻辑回归模型来研究睡眠问题对儿童和家庭的影响与年龄组、突变类型、药物类型以及睡眠卫生评分之间的关系。使用线性回归模型来估计入睡和维持睡眠障碍(DIMS)与年龄组、突变类型、药物类型以及睡眠卫生之间的关联。在那些曾经有入睡困难或夜间醒来问题的人群中,与未治疗/未用药组相比,在考虑年龄、突变类型和睡眠卫生评分影响的情况下,使用任何药物都与受影响儿童和家庭出现中度/重度影响睡眠问题(相对于轻度/无影响)的较高几率相关,同时DIMS评分也更高。与较差的睡眠卫生习惯使用情况相比,在调整协变量后,更好地运用睡眠卫生习惯与对家庭产生中度/重度影响的较低几率(优势比0.60,95%置信区间[CI] 0.37,0.98)以及较低的DIMS评分(几何平均比0.86,95%CI 0.80,0.92)相关。关注睡眠卫生仍然是RTT睡眠问题的一项重要管理策略。需要进一步开展前瞻性研究来调查药物治疗的疗效。

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