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发作性睡病/猝倒症儿童及青少年的睡眠多导生理研究

Sleep polygraphic study of children and adolescents with narcolepsy/cataplexy.

作者信息

Ferri Raffaele, Franceschini Christian, Zucconi Marco, Drago Valeria, Manconi Mauro, Vandi Stefano, Poli Francesca, Bruni Oliviero, Plazzi Giuseppe

机构信息

Sleep Research Centre, Department of Neurology I.C., Oasi Institute (IRCCS), Troina, Italy.

出版信息

Dev Neuropsychol. 2009;34(5):523-38. doi: 10.1080/87565640903133699.

Abstract

The alterations of the Cyclic Alternating Pattern (CAP) recently found in narcoleptic adult patients suggest the presence of an impaired modulation of the fluctuations of the arousal level during their non-rapid eye movement (NREM) sleep, possibly because of the persistence of neurophysiological mechanisms typical of rapid eye movement (REM) sleep. The same mechanism might play a role in the occurrence of leg movement (LM) activity during sleep characterized by low levels of periodicity. The aim of this study was to evaluate CAP and sleep LM activity in a group of children and adolescents with narcolepsy, to interpret the results under a developmental point of view and integrate this new information with data already available for adults. Thirteen young patients with narcolepsy/cataplexy were consecutively recruited for this study, together with 13 age- and sex-matched normal controls. Nocturnal polysomnography was carried out after a night of adaptation in a sleep laboratory room; sleep stages, CAP, and LMs were scored and evaluated following standard criteria. Narcoleptic patients showed shorter sleep onset and REM sleep latency, higher number of stage shifts and awakenings per hour of sleep, and higher percentage of wakefulness after sleep onset; CAP rate was found to be decreased in all NREM sleep stages (in particular CAP A1 subtypes) in narcoleptic patients who also showed significant higher values of all types of LMs (periodic or isolated), during both REM and NREM sleep; however, the most evident differences were found during REM sleep. The results of this study confirm that the sleep microstructure and LM activity changes observed in adulthood are already present and detectable in childhood and might have a role in the already known impaired prefrontal functioning of these subjects. The well-established orexin deficiency might be the unifying factor playing a major role in the modulation of CAP and LMs during sleep in children and adolescents with narcolepsy/cataplexy.

摘要

最近在发作性睡病成年患者中发现的周期性交替模式(CAP)改变表明,在他们的非快速眼动(NREM)睡眠期间,觉醒水平波动的调节受损,这可能是由于快速眼动(REM)睡眠典型的神经生理机制持续存在所致。相同的机制可能在睡眠期间周期性水平较低的腿部运动(LM)活动的发生中起作用。本研究的目的是评估一组发作性睡病儿童和青少年的CAP和睡眠LM活动,从发育的角度解释结果,并将这些新信息与已有的成人数据相结合。本研究连续招募了13名患有发作性睡病/猝倒症的年轻患者,以及13名年龄和性别匹配的正常对照。在睡眠实验室房间适应一晚后进行夜间多导睡眠图检查;按照标准标准对睡眠阶段、CAP和LM进行评分和评估。发作性睡病患者的睡眠起始和REM睡眠潜伏期较短,每小时睡眠中的阶段转换和觉醒次数较多,睡眠起始后的清醒百分比较高;在所有NREM睡眠阶段(特别是CAP A1亚型),发作性睡病患者的CAP率均降低,这些患者在REM和NREM睡眠期间还表现出所有类型LM(周期性或孤立性)的显著更高值;然而,最明显的差异出现在REM睡眠期间。本研究结果证实,在成年期观察到的睡眠微观结构和LM活动变化在儿童期已经存在且可检测到,并且可能在这些受试者已知的前额叶功能受损中起作用。公认的食欲素缺乏可能是在发作性睡病/猝倒症儿童和青少年睡眠期间调节CAP和LM中起主要作用的统一因素。

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