PDF(Pubmed)
Abstract：
BACKGROUND: There is a critical need for the development of improved outcome measures in Fragile X Syndrome (FXS). Because the majority of respondents of behavior outcome measures are caregivers or individuals with FXS, it is important to consider stakeholders\' firsthand experiences when designing a caregiver- or self-report measure.
OBJECTIVE: The current research study aimed to understand experiences of completing commonly used caregiver-/self-report measures of behavior in FXS via focus groups.
METHODS: This study employed a focus group methodology. Semi-structured focus groups were conducted with 22 caregivers and 3 self-advocates. All interviews occurred via secured videoconferencing. A thematic analysis was used to identify major themes and subthemes.
RESULTS: We identified four themes: (1) content of measure, (2) structure of the measure, (3) potential accommodations to complete measure, and (4) impact of measure on family. Importantly, focus groups revealed that certain aspects of content, structure, and implementation of the available measures were related to distress and negative emotions of caregivers of FXS and individuals with FXS themselves.
CONCLUSIONS: The focus group data yielded a wide range of feedback and has significant implications, highlighting the critical need to take key stakeholder perspectives into account when using and/or developing caregiver- or self-report measures for FXS.
OBJECTIVE: The current research study aimed to understand experiences of completing commonly used caregiver-/self-report measures of behavior in FXS via focus groups.
METHODS: This study employed a focus group methodology. Semi-structured focus groups were conducted with 22 caregivers and 3 self-advocates. All interviews occurred via secured videoconferencing. A thematic analysis was used to identify major themes and subthemes.
RESULTS: We identified four themes: (1) content of measure, (2) structure of the measure, (3) potential accommodations to complete measure, and (4) impact of measure on family. Importantly, focus groups revealed that certain aspects of content, structure, and implementation of the available measures were related to distress and negative emotions of caregivers of FXS and individuals with FXS themselves.
CONCLUSIONS: The focus group data yielded a wide range of feedback and has significant implications, highlighting the critical need to take key stakeholder perspectives into account when using and/or developing caregiver- or self-report measures for FXS.
摘要：
背景：迫切需要开发脆性X综合征（FXS）的改善结果指标。因为行为结果测量的大多数受访者是护理人员或FXS患者，在设计照顾者或自我报告措施时，考虑利益相关者的第一手经验是很重要的。
目的：当前的研究旨在了解通过焦点小组完成FXS中常用的照顾者/自我报告行为测量的经验。
方法：本研究采用焦点小组方法。半结构化焦点小组由22名护理人员和3名自我倡导者组成。所有采访都是通过安全的视频会议进行的。专题分析用于确定主要主题和次主题。
结果：我们确定了四个主题：（1）度量内容，(2)措施的结构，(3)完成测量的潜在调节，(4)措施对家庭的影响。重要的是,焦点小组透露，内容的某些方面，结构,现有措施的实施与FXS护理人员和FXS患者自身的痛苦和负面情绪有关。
结论：焦点小组数据产生了广泛的反馈，具有重要意义，强调在使用和/或开发FXS的护理人员或自我报告措施时，必须考虑关键利益相关者的观点。
目的：当前的研究旨在了解通过焦点小组完成FXS中常用的照顾者/自我报告行为测量的经验。
方法：本研究采用焦点小组方法。半结构化焦点小组由22名护理人员和3名自我倡导者组成。所有采访都是通过安全的视频会议进行的。专题分析用于确定主要主题和次主题。
结果：我们确定了四个主题：（1）度量内容，(2)措施的结构，(3)完成测量的潜在调节，(4)措施对家庭的影响。重要的是,焦点小组透露，内容的某些方面，结构,现有措施的实施与FXS护理人员和FXS患者自身的痛苦和负面情绪有关。
结论：焦点小组数据产生了广泛的反馈，具有重要意义，强调在使用和/或开发FXS的护理人员或自我报告措施时，必须考虑关键利益相关者的观点。
