Developing improved outcome measures in FXS: Key stakeholder feedback.
Res Dev Disabil
; 137: 104502, 2023 Jun.
Article
em En
| MEDLINE
| ID: mdl-37080087
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 ANDRESULTS:
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.Palavras-chave
Texto completo:
1
Base de dados:
MEDLINE
Assunto principal:
Síndrome do Cromossomo X Frágil
Tipo de estudo:
Qualitative_research
Limite:
Humans
Idioma:
En
Ano de publicação:
2023
Tipo de documento:
Article