Study Objectives:

Excessive daytime sleepiness is common in Prader-Willi Syndrome (PWS), with prevalence ranging from 52%-100%. The goal of this study was to establish the content validity (i.e., evidence that an instrument measures an intended concept of interest) of the parent/caregiver version of the Epworth Sleepiness Scale for Children and Adolescents (ESS-CHAD), a measure of daytime sleepiness, in PWS.

Methods:

Qualitative, dyadic semi-structured video interviews were conducted with 18 caregivers and their children with PWS from April-June 2020. Concept elicitation and cognitive interview techniques were implemented. Thematic analyses allowed for examination of themes and data patterns.

Results:

All caregivers (mean age 49 years) were mothers of individuals with PWS who experienced troublesome daytime sleepiness (mean age 14 years). The most prevalent observable signs/symptoms of daytime sleepiness were: sleepy/sleepiness (n=17; 94.4%), tired/tiredness (n=16; 88.9%), exhaustion/exhausted (n=5; 27.8%), anxious/stressed (n=5; 27.8%), irritable/frustrated (n=5; 27.8%), having tantrums/outbursts (n=5; 27.8%), and lethargy (n=4; 22.2%). Daytime sleepiness impacted various aspects of health including mental, emotional, physical, and social well-being. When caregivers were asked about the activities associated with daytime sleepiness, all salient concepts elicited mapped to the ESS-CHAD; saturation was met after the first four interviews. Only two concepts, after physical exertion and while inactive/bored, did not map. Caregiver statements indicated that these concepts, although related to daytime activities, were atypical of daily routines. The ESS-CHAD was well understood and relevant to caregivers.

Conclusions:

This study supports the content validity of the ESS-CHAD and its appropriateness for evaluating treatment efficacy of daytime sleepiness in PWS.

中文翻译：

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建立普瑞德威利综合征儿童和青少年 Epworth 嗜睡量表的内容效度

学习目标：

白天过度嗜睡在普瑞德威利综合症 (PWS) 中很常见，患病率范围为 52%-100%。本研究的目的是确定儿童和青少年 Epworth 嗜睡量表 (ESS-CHAD) 的家长/看护者版本的内容有效性（即，仪器测量预期兴趣概念的证据），该量表是一种白天的测量方法。嗜睡，PWS。

方法：

自 2020 年 4 月至 6 月，我们对 18 名患有 PWS 的护理人员及其孩子进行了定性、二元半结构化视频访谈。采用了概念启发和认知访谈技术。主题分析允许检查主题和数据模式。

结果：

所有看护者（平均年龄 49 岁）都是 PWS 患者的母亲，她们经历过日间嗜睡（平均年龄 14 岁）。白天嗜睡最常见的可观察体征/症状是：困倦/困倦 (n=17; 94.4%)、疲倦/疲倦 (n=16; 88.9%)、精疲力竭/疲惫 (n=5; 27.8%)、焦虑/压力大 (n=5; 27.8%)、烦躁/沮丧 (n=5; 27.8%)、发脾气/爆发 (n=5; 27.8%) 和嗜睡 (n=4; 22.2%)。白天嗜睡影响健康的各个方面，包括心理、情感、身体和社交福祉。当护理人员被问及与白天嗜睡相关的活动时，所有显着概念都被引出到 ESS-CHAD；前四次面试后就达到了饱和。只有两个概念，在体力消耗后和不活动/无聊时没有映射。护理人员的陈述表明，这些概念虽然与白天活动有关，但却是日常生活中的非典型情况。ESS-CHAD 易于理解并且与护理人员相关。

结论：

本研究支持 ESS-CHAD 的内容有效性及其评估 PWS 日间嗜睡治疗效果的适当性。