Department of Biomedical Sciences and Engineering, National Central University, Taoyuan, Taiwan.
Division of Pediatric Psychiatry and Sleep Center, Chang Gung Memorial Hospital, Taoyuan, Taiwan.
Sleep. 2021 Sep 13;44(9). doi: 10.1093/sleep/zsab096.
Kleine-Levin syndrome (KLS) is a rare recurrent hypersomnia. Our study aimed at monitoring the movements of patients with KLS using actigraphy and evaluating their circadian rhythm.
Twenty young patients with KLS and 14 age-matched controls were recruited. Each individual wore an actigraphy for more than 6 months to monitor at least two attacks. Controls kept wearing the device for at least 7 days. The activity counts were averaged in hourly basis and the day-to-night amplitude was quantified by the differences of the averaged activity counts during daytime and nighttime. The hourly activities of different days were aligned and averaged to construct the circadian profile. Parametric and nonparametric estimation of circadian rhythm was calculated. We applied detrended fluctuation analysis to evaluate the temporal correlations beneath the activity fluctuations at multiple time scales.
Circadian rhythm in asymptomatic period showed no significant difference compared to the controls. During hypersomnia attack, the amplitude of the circadian rest-active rhythms drastically decreased and decreased interdaily stability (IS) was found, as well as significant decreased M10 and short-time fractal correlation (α1). Drastically decreased mean and standard deviation of activity were noted, compared to the pre-attack phase and recovery phase. α1 and M10 increased during the late attack phase, and overcompensated IS was noted in the recovery phase.
This study confirmed that circadian rest-active rhythms was affected when KLS hypersomnia attack. Several parameters including M10, IS, and α1 may be physiological markers of KLS, which can help to predict the end of hypersomnia episodes.
克莱恩-莱文综合征（KLS）是一种罕见的反复发作性嗜睡症。本研究旨在通过活动记录仪监测 KLS 患者的运动，并评估其昼夜节律。
招募了 20 名 KLS 年轻患者和 14 名年龄匹配的对照组。每位个体佩戴活动记录仪超过 6 个月，以监测至少两次发作。对照组至少佩戴设备 7 天。以每小时为基础平均活动计数，并通过白天和夜间平均活动计数之间的差异量化昼夜幅度。对齐不同天数的每小时活动并进行平均，以构建昼夜节律曲线。计算昼夜节律的参数和非参数估计。我们应用去趋势波动分析来评估在多个时间尺度下活动波动下的时间相关性。
无症状期的昼夜节律与对照组相比无显著差异。在嗜睡发作期间，昼夜休息-活动节律的幅度明显减小，日间稳定性（IS）降低，以及明显降低的 M10 和短时间分形相关性（α1）。与发作前阶段和恢复期相比，活动的平均值和标准差明显降低。在发作后期，α1 和 M10 增加，恢复期出现过度补偿的 IS。
本研究证实，KLS 嗜睡发作时昼夜休息-活动节律受到影响。包括 M10、IS 和α1 在内的几个参数可能是 KLS 的生理标志物，有助于预测嗜睡发作的结束。
