Developing improved outcome measures in FXS: Key stakeholder feedback

• Published in: Research in developmental disabilities 2023-06, Vol.137, p.104502-104502, Article 104502
• Main Authors: Kang, Sungeun, Jones, Angelina, Shaffer, Rebecca C., Erickson, Craig A., Schmitt, Lauren M.
• Format: Article
• Language: English
• Subjects: Caregiver perspectives; Caregivers - psychology; Feedback; Fragile X Syndrome; Fragile X Syndrome - psychology; Humans; Individuals with developmental disabilities; Outcome Assessment, Health Care; Outcome Measure; Self Report; Stakeholder; Voice of the Patient
• ISSN: 0891-4222; 1873-3379
• DOI: 10.1016/j.ridd.2023.104502

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. •Caregiver- and self-report measures have limitations in Fragile X Syndrome.•We conducted focus groups to gather key stakeholders' experiences.•The content and structure of the measure are important.•Caregiver-report measures may cause distress and negative emotions in respondents.•Stakeholders' experiences are critical when developing an outcome measure.