The objectives of this study were to characterize the sleep habits of 50 clinically referred individuals with Angelman syndrome (AS) and to retrospectively compare the effectiveness/tolerability of the three most commonly prescribed sleep medications in the sample. An experienced physician assigned a Clinical Global Impressions‐Severity scale (CGI‐S) score for each subject's AS‐specific symptoms. Caregivers completed the Child Sleep Habits Questionnaire (CSHQ; screen for sleep problems in school‐aged [4–10 years] children), a screening assessment for sleep problems. Caregivers provided information about medication trials targeting disturbed sleep, with the physician assigning a CGI‐Improvement scale (CGI‐I) score for each trial. Linear regression showed significant negative association between age and CSHQ score. In their lifetime, 72% of participants had taken a medication for sleep, most commonly melatonin, clonidine and trazodone. The majority continued these for 6 months or longer. With these medications, many demonstrated significant improvement in sleep disturbances, with no difference in odds of improvement between medications. Disturbed sleep was common in this cohort and significantly worse in younger‐aged participants. The majority received at least one medication trial for disturbed sleep and each of the most commonly prescribed medication was effective for a substantial percentage of participants. Most participants remained on medication for at least 6 months, suggesting favorable tolerability.

中文翻译：

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患有Angelman综合征的儿童和成人的睡眠习惯和药物治疗对睡眠障碍的影响。

这项研究的目的是表征50名临床上有Angelman综合征（AS）的临床转诊患者的睡眠习惯，并回顾性比较样本中三种最常用处方睡眠药物的有效性/耐受性。一位经验丰富的医生为每个受试者的AS特定症状分配了临床总体印象-严重程度量表（CGI-S）评分。护理人员完成了“儿童睡眠习惯问卷”（CSHQ；对学龄[4-10岁]儿童的睡眠问题进行筛查），这是一项针对睡眠问题的筛查评估。护理人员提供了有关针对睡眠障碍的药物试验的信息，医生为每个试验分配了CGI改善量表（CGI-I）评分。线性回归显示年龄与CSHQ得分之间显着负相关。在他们的一生中，72％的参与者服用了睡眠药物，最常见的是褪黑激素，可乐定和曲唑酮。大多数人将这些持续了6个月或更长时间。使用这些药物，许多人表现出睡眠障碍的显着改善，而两种药物之间改善的几率没有差异。睡眠障碍在该队列中很常见，而在较年轻的参与者中则明显恶化。大多数人至少接受了一项针对睡眠不安的药物试验，并且每种最常用的药物对相当多的参与者有效。大多数参与者服药至少6个月，表明耐受性良好。许多人表现出睡眠障碍的显着改善，两种药物之间改善的几率没有差异。睡眠障碍在该队列中很常见，而在较年轻的参与者中则明显恶化。大多数人至少接受了一项针对睡眠不安的药物试验，并且每种最常用的药物对相当多的参与者有效。大多数参与者服药至少6个月，表明耐受性良好。许多人表现出睡眠障碍的显着改善，两种药物之间改善的几率没有差异。睡眠障碍在该队列中很常见，而在较年轻的参与者中则明显恶化。大多数人至少接受了一项针对睡眠不安的药物试验，并且每种最常用的药物对相当多的参与者有效。大多数参与者服药至少6个月，表明耐受性良好。大多数人至少接受了一项针对睡眠不安的药物试验，并且每种最常用的药物对相当多的参与者有效。大多数参与者服药至少6个月，表明耐受性良好。大多数人至少接受了一项针对睡眠不安的药物试验，并且每种最常用的药物对相当多的参与者有效。大多数参与者服药至少6个月，表明耐受性良好。