Developing a Down Syndrome Health Instrument
试验速览
- 阶段
- 不适用
- 状态
- 已完成
- 入组人数
- 758
- 试验地点
- 1
- 主要终点
- Survey results, cognitive interviews, and validation
研究概览
简要总结
Although over 200,000 individuals with DS live in the United States, studies to date have focused on outcomes apart from health. The foundation for this proposal is based on the need to accurately measure health of all individuals - specifically, with DS - and the dearth of available tools for this population. Creating such an instrument will provide a barometer of the current state of health for DS and hold use in future research. In this project, I propose to create an instrument that directly assesses health in DS - the Down syndrome Health Instrument (DHI). More specifically, the aims of this proposal are: 1. To conduct focus groups among caregivers, individuals with DS, panels of experts on DS and primary care physicians, and cognitive interviews to refine a conceptual model of health for DS and create an item pool, 2. To administer the DHI and establish internal validity, reliability, and external validity of the DHI for use in clinical research, and 3. To test the usability of the DHI in two pilot settings: research and clinical. This instrument will measure patient-reported health in DS for the first time and allow measurement of health as an outcome which is not currently possible in this population. This can identify gaps in care, then direct and optimize interventions that will improve care.
研究设计
- 研究类型
- Observational
- 观察模型
- Cohort
- 时间视角
- Prospective
入排标准
- 年龄范围
- 18 Years 至 —(Adult, Older Adult)
- 性别
- All
- 接受健康志愿者
- 否
入选标准
- •Primary caregiver of an individual with DS (individual with DS age: <22 years)
- •Caregiver age: ≥18 years
- •Fluent in written and spoken English
- •Able to read and provide informed consent
排除标准
- •Physical or mental condition of caregiver that would prohibit self-administration of questionnaire
- •Mosaic Down syndrome: based on medical record review. If caregiver is uncertain or mosaicism is unknown, we will request additional records.
研究组 & 干预措施
Focus Groups
N=52
干预措施: Observational, no intervention (Other)
Cognitive Interviews
N=24
干预措施: Observational, no intervention (Other)
Survey
N=542
干预措施: Observational, no intervention (Other)
结局指标
主要结局
Survey results, cognitive interviews, and validation
时间窗: Within 12 months of completion of analysis of surveys
Local and national survey results, cognitive interviews and validation of DHI
Number of Completed Surveys for Validation Analysis
时间窗: At the time of survey completion
Local and national survey results, cognitive interviews and validation of Down syndrome health measure (DSHM) using psychometric analysis and factor analysis
次要结局
- Focus group results and conceptual model(Within 12 months of completion of focus groups)
- Number of Focus Group Participants Who Participated to Make the Conceptual Model(At the time of focus group completion)
- Number of Completed Cognitive Interviews(At the time of cognitive interview)
研究者
Stephanie Santoro
Assistant Professor of Pediatrics, Clinical Geneticist
Massachusetts General Hospital
