Abstract：
OBJECTIVE: Sleep difficulties are common in CDKL5 deficiency disorder (CDD), a developmental and epileptic encephalopathy (DEE). This study evaluated the factor structure of the Disorders of Initiating and Maintaining Sleep (DIMS), Disorders of Excessive Daytime Somnolence (DOES) and Sleep Breathing Disorders (SBD) domains of the Sleep Disturbance Scale for Children (SDSC) for CDD.
METHODS: A cross-sectional psychometric study design was used. Data were collected for 125 individuals aged 3 years or older who attended a US Centers of Excellence clinic or registered with the International CDKL5 Disorder Database.
RESULTS: The median age was 10.3 years (range 3.2 - 40.7 years) and 105 (84%) were female. Two of the three SBD items related were not observed by most respondents and analysis was restricted to the DIMS and DOES domains. Using all items in the initial confirmatory factor analysis, two items in the DIMS domain and one item in the DOES domain loaded poorly. After deleting these items and repeating the analysis, item loading (0.524-0.814) and internal consistency (DIMS: 0.78, DOES: 0.76) statistics were good. The square of the inter-domain correlation coefficient was 0.17, less than Average Variance Extracted values for both domains and indicating good discriminant validity. The Tucker-Lewis and Comparative Fit indices were slightly lower than the threshold of >0.9 for establishing goodness of fit.
CONCLUSIONS: The modified DIMS and DOES domains from the SDSC could be suitable clinical outcome assessments of insomnia and related impairments in CDD and potentially other DEE conditions.
METHODS: A cross-sectional psychometric study design was used. Data were collected for 125 individuals aged 3 years or older who attended a US Centers of Excellence clinic or registered with the International CDKL5 Disorder Database.
RESULTS: The median age was 10.3 years (range 3.2 - 40.7 years) and 105 (84%) were female. Two of the three SBD items related were not observed by most respondents and analysis was restricted to the DIMS and DOES domains. Using all items in the initial confirmatory factor analysis, two items in the DIMS domain and one item in the DOES domain loaded poorly. After deleting these items and repeating the analysis, item loading (0.524-0.814) and internal consistency (DIMS: 0.78, DOES: 0.76) statistics were good. The square of the inter-domain correlation coefficient was 0.17, less than Average Variance Extracted values for both domains and indicating good discriminant validity. The Tucker-Lewis and Comparative Fit indices were slightly lower than the threshold of >0.9 for establishing goodness of fit.
CONCLUSIONS: The modified DIMS and DOES domains from the SDSC could be suitable clinical outcome assessments of insomnia and related impairments in CDD and potentially other DEE conditions.
摘要：
目的：睡眠困难在CDKL5缺乏症（CDD）中很常见，发育性和癫痫性脑病（DEE）。这项研究评估了启动和维持睡眠障碍（DIMS）的因素结构，CDD儿童睡眠障碍量表（SDSC）的日间睡眠过度障碍（DOES）和睡眠呼吸障碍（SBD）领域。
方法：采用横断面心理测量学研究设计。收集了125名年龄在3岁或以上的人的数据，他们参加了美国卓越中心诊所或在国际CDKL5疾病数据库注册。
结果：中位年龄为10.3岁（范围3.2-40.7岁），女性为105岁（84%）。大多数受访者未观察到三个SBD项目中的两个，并且分析仅限于DIMS和DOES领域。使用初始验证性因子分析中的所有项目，DIMS域中的两个项目和DOES域中的一个项目加载不良。删除这些项目并重复分析后，项目加载（0.524-0.814）和内部一致性（DIMS：0.78，DOES：0.76）统计数据良好。域间相关系数的平方为0.17，小于两个域的平均方差提取值，表明判别有效性良好。Tucker-Lewis和比较拟合指数略低于>0.9的阈值以建立拟合优度。
结论：来自SDSC的改良的DIMS和DOES域可能是CDD和潜在的其他DEE病症中失眠和相关损伤的合适临床结果评估。
方法：采用横断面心理测量学研究设计。收集了125名年龄在3岁或以上的人的数据，他们参加了美国卓越中心诊所或在国际CDKL5疾病数据库注册。
结果：中位年龄为10.3岁（范围3.2-40.7岁），女性为105岁（84%）。大多数受访者未观察到三个SBD项目中的两个，并且分析仅限于DIMS和DOES领域。使用初始验证性因子分析中的所有项目，DIMS域中的两个项目和DOES域中的一个项目加载不良。删除这些项目并重复分析后，项目加载（0.524-0.814）和内部一致性（DIMS：0.78，DOES：0.76）统计数据良好。域间相关系数的平方为0.17，小于两个域的平均方差提取值，表明判别有效性良好。Tucker-Lewis和比较拟合指数略低于>0.9的阈值以建立拟合优度。
结论：来自SDSC的改良的DIMS和DOES域可能是CDD和潜在的其他DEE病症中失眠和相关损伤的合适临床结果评估。
