Agar Georgie, Oliver Chris, Spiller Jayne, Richards Caroline
School of Psychology, Aston University, Birmingham, UK.
School of Psychology, University of Birmingham, Birmingham, UK.
Sleep Adv. 2023 Sep 8;4(1):zpad034. doi: 10.1093/sleepadvances/zpad034. eCollection 2023.
To determine the trajectory of: (i) objective sleep parameters and (ii) caregiver-reported sleep questionnaire scores over 3 years in children with Smith-Magenis syndrome (SMS) compared to age-matched typically developing (TD) controls. We also aimed to (iii) describe individual profiles of change in sleep parameters over time.
Week-long, overnight actigraphy and questionnaire data from 13 children with SMS and 13 age-matched TD children were collected at Time 1 and Time 2 (3 years later). Independent samples -tests, paired samples -tests, and Bayesian analyses were used to compare sleep parameters and sleep questionnaire scores between groups at each time point and compare data within groups to assess change over time.
Sleep parameters were consistently more disrupted in the SMS group than the TD group, with significantly reduced sleep efficiency, increased wake after sleep onset and earlier get up times at both time points. This was mirrored in the questionnaire data, with children with SMS evidencing higher scores for overall sleep disturbance, night waking, and daytime sleepiness. While TD sleep parameters demonstrated expected developmental changes over 3 years, in the SMS group sleep parameters and variability between and within children remained largely stable. However, some children with SMS showed substantial variation in sleep parameters over time. Questionnaire scores remained stable over 3 years in both groups.
Overall, sleep disturbance appears to be a stable feature of SMS, indicative of a divergent sleep trajectory compared to TD peers. Proactive intervention approaches should be considered for poor sleep in SMS.
确定史密斯-马吉尼斯综合征（SMS）患儿与年龄匹配的正常发育（TD）对照儿童相比，在3年时间里：（i）客观睡眠参数和（ii）照料者报告的睡眠问卷得分的变化轨迹。我们还旨在（iii）描述睡眠参数随时间变化的个体特征。
在第1时间点和第2时间点（3年后）收集了13名SMS患儿和13名年龄匹配的TD儿童为期一周的夜间活动记录仪数据和问卷数据。使用独立样本t检验、配对样本t检验和贝叶斯分析来比较各时间点两组之间的睡眠参数和睡眠问卷得分，并比较组内数据以评估随时间的变化。
SMS组的睡眠参数始终比TD组更紊乱，在两个时间点睡眠效率均显著降低，睡眠开始后的觉醒增加，起床时间更早。问卷数据也反映了这一点，SMS患儿在总体睡眠障碍、夜间觉醒和白天嗜睡方面得分更高。虽然TD组的睡眠参数在3年中呈现出预期的发育变化，但SMS组儿童的睡眠参数以及儿童之间和儿童内部的变异性在很大程度上保持稳定。然而，一些SMS患儿的睡眠参数随时间有显著变化。两组的问卷得分在3年中均保持稳定。
总体而言，睡眠障碍似乎是SMS的一个稳定特征，表明与TD同龄人相比睡眠轨迹不同。对于SMS患儿的睡眠不佳应考虑采取积极的干预措施。
