ArticleResearch in developmental disabilities2023
Developing improved outcome measures in FXS: Key stakeholder feedback.
Article in Research in developmental disabilities, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
1 citing paper in PubMed, 1 citations in OpenAlex.
- Navigating the outcome maze: a scoping review of outcomes and instruments in clinical trials in genetic neurodevelopmental disorders and intellectual disability.Therapeutic advances in rare diseaseArticle
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
5 authors at 2 institutions in 1 country.
Funding
Abstract
backgroundThere 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.
aimsThe 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
resultsWe 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.
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Registered trials
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