Developing improved outcome measures in FXS: Key stakeholder feedback.
Current FXS caregiver forms cause distress; co-design new ones or expect shaky data.
01Research in Context
What this study did
Kang et al. (2023) ran online focus groups. They asked caregivers and adults with fragile X what they think of current behavior forms.
The team wanted to know why the forms feel hard, upsetting, or wrong.
What they found
People said the questions are confusing, too long, and stir up worry or guilt.
They want shorter forms, kinder words, and a say in how the tool is used.
How this fits with other research
Lemons et al. (2015) looked at 31 small FXS studies and said the evidence is thin. Sungeun’s work shows one reason: the tools themselves stress families, so data quality drops.
Montanaro et al. (2024) ran a year-long group therapy for adults with FXS and used the same caregiver forms Sungeun now critiques. The positive gains they report might be under-cut by rater fatigue or bias.
Klein et al. (2024) found that stressed caregivers give skewed quality-of-life scores. Sungeun’s groups echo this: upset caregivers fill out forms differently.
Why it matters
Before you pick an FXS outcome tool, ask: will this form upset the person filling it out? Try a short pilot with two caregivers. Note where they sigh, pause, or look worried. Swap those items for plain, friendly language. Happy caregivers give cleaner data.
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02At a glance
03Original 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. AIMS: The current research study aimed to understand experiences of completing commonly used caregiver-/self-report measures of behavior in FXS via focus groups. METHODS AND PROCEDURES: 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. OUTCOMES AND 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 AND IMPLICATIONS: 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.
Research in developmental disabilities, 2023 · doi:10.3389/fpsyg.2023.1118652