Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA.
Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA; University of Cincinnati College of Medicine, Cincinnati, OH, USA.
Res Dev Disabil. 2023 Jun;137:104502. doi: 10.1016/j.ridd.2023.104502. Epub 2023 Apr 18.
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.
The current research study aimed to understand experiences of completing commonly used caregiver-/self-report measures of behavior in FXS via focus groups.
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.
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.
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.
脆性 X 综合征（FXS）需要开发改进的预后评估方法。由于行为预后评估的大多数受访者是照顾者或 FXS 患者，因此在设计照顾者或自我报告评估工具时，考虑利益相关者的第一手经验非常重要。
本研究旨在通过焦点小组了解 FXS 中常用的照顾者/自我报告行为评估工具的使用体验。
本研究采用焦点小组方法。对 22 名照顾者和 3 名自我倡导者进行了半结构化焦点小组访谈。所有访谈均通过安全视频会议进行。采用主题分析法确定主要主题和子主题。
我们确定了四个主题：（1）评估工具的内容；（2）评估工具的结构；（3）完成评估工具的潜在调整；（4）评估工具对家庭的影响。重要的是，焦点小组揭示了现有评估工具的内容、结构和实施的某些方面与 FXS 照顾者和患者的困扰和负面情绪有关。
焦点小组数据提供了广泛的反馈，具有重要意义，强调在使用和/或开发 FXS 的照顾者/自我报告评估工具时，需要考虑关键利益相关者的观点。
