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Uncovering Sleep in Young Males with Duchenne Muscular Dystrophy
European Journal of Paediatric Neurology ( IF 3.1 ) Pub Date : 2020-05-01 , DOI: 10.1016/j.ejpn.2020.02.012
Amy G Hartman 1 , Lauren Terhorst 2 , Natalie Little 1 , Roxanna M Bendixen 1
Affiliation  

OBJECTIVES Sleep health in rare disease is often overlooked due to the complex nature of the disease. For males with Duchenne muscular dystrophy, sleep assessment is typically focused on pulmonary function and identification of sleep disordered breathing. Unfortunately for young boys with Duchenne muscular dystrophy, sleep assessment is often neglected, resulting in a dearth of knowledge on sleep health in this population. This study describes sleep quantity and quality in both younger (4-9 years) and older (10-17 years) males with Duchenne muscular dystrophy (n = 19) and compares these characteristics with sleep characteristics of unaffected peers (n = 17). METHODS This study was a longitudinal, observational study. Sleep measures were collected using the parent-proxy Children's Sleep Habits Questionnaire-Abbreviated version and objective sleep measures from actigraphy (sleep efficiency, awakenings, and awakening duration) over 30 days for all participants. Means and standard deviations were examined, and effect sizes were computed to quantify the magnitude of difference between the Duchenne muscular dystrophy and unaffected groups. RESULTS Overall, boys with Duchenne muscular dystrophy were found to experience worse sleep than their unaffected peers as measured by parent report and actigraphy. Effect sizes of both measures demonstrated moderate to large magnitudes of difference in many of the sleep variables. Parents of boys with Duchenne muscular dystrophy reported higher scores (indicating worse sleep) in all subsections and total score of the Children's Sleep Habits Questionnaire - Abbreviated version. Actigraphy data indicated that the Duchenne muscular dystrophy group had lower percent sleep efficiency, more night awakenings and longer duration of night awakenings than their unaffected peers. CONCLUSION Our findings offer a novel look into sleep in young boys with Duchenne muscular dystrophy. Both parent-report and actigraphy data indicate poor sleep health in this population compared with age-matched unaffected peers. Actigraphy was found to align with parent-report of sleep in this population, supporting the use of these two different ways to measure sleep in Duchenne muscular dystrophy. Results from this study should encourage clinicians and researchers alike to further explore sleep and its impact on disease in young boys with Duchenne muscular dystrophy.

中文翻译:

揭示患有杜氏肌营养不良症的年轻男性的睡眠

目标 由于疾病的复杂性,罕见病患者的睡眠健康常常被忽视。对于患有杜氏肌营养不良症的男性,睡眠评估通常侧重于肺功能和睡眠呼吸障碍的识别。不幸的是,对于患有杜氏肌营养不良症的小男孩来说,睡眠评估往往被忽视,导致该人群缺乏关于睡眠健康的知识。本研究描述了患有杜氏肌营养不良症的年轻(4-9 岁)和年长(10-17 岁)男性(n = 19)的睡眠数量和质量,并将这些特征与未受影响的同龄人(n = 17)的睡眠特征进行了比较。方法 本研究是一项纵向观察性研究。睡眠测量是使用父母代理儿童收集的 s 睡眠习惯问卷——所有参与者超过 30 天的活动记录(睡眠效率、觉醒和觉醒持续时间)的缩写版本和客观睡眠测量。检查了平均值和标准差,并计算了效应量以量化杜氏肌营养不良症和未受影响组之间的差异程度。结果 总体而言,根据父母报告和活动记录的测量,发现患有杜氏肌营养不良症的男孩比未受影响的同龄人的睡眠更差。两种测量的效果大小在许多睡眠变量中都显示出中到大的差异。Duchenne 型肌营养不良症男孩的父母在儿童睡眠习惯问卷 - 缩略版的所有小节和总分中报告了较高的分数(表明睡眠较差)。活动记录数据表明,与未受影响的同龄人相比,杜氏肌营养不良症组的睡眠效率百分比较低,夜间觉醒次数更多,夜间觉醒持续时间更长。结论 我们的研究结果为患有杜氏肌营养不良症的年轻男孩的睡眠提供了一种新的视角。父母报告和活动记录数据均表明,与年龄匹配的未受影响同龄人相比,该人群的睡眠健康状况较差。活动记录被发现与该人群的父母睡眠报告一致,支持使用这两种不同的方法来测量杜氏肌营养不良症的睡眠。这项研究的结果应该鼓励临床医生和研究人员进一步探索睡眠及其对杜氏肌营养不良症年轻男孩疾病的影响。
更新日期:2020-05-01
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