背景:患有和不患有唐氏综合症(DS)的人的招募挑战会延迟研究进展和风险样本偏差。这项研究确定并量化了具有和不具有DS的个人的研究招募决策者人群的研究态度差异。
方法:我们使用来自两个注册管理机构的数据进行分析:加州大学,IrvineConsent-to-Contact(C2C)RegistryandDS-Connect.前者代表非DS决策者的样本(N=4818),而对于后者,我们排除了DS患者,留下一群DS家庭决策者(N=976)。我们评估了DS和非DS决策者之间的研究态度问卷(RAQ)的得分。我们使用线性回归比较总RAQ评分,并使用比例赔率回归评估项目水平RAQ差异。
结果:两个注册管理机构的决策者之间的平均总RAQ评分没有统计学差异,在调整了年龄之后,性别,种族和民族,教育,和2019年冠状病毒病(COVID-19)时间框架(Est。Diff=0.11,95%置信区间[CI]:-0.22,0.43;p=0.531)。然而,在预先指定的分析中,我们确实发现了对项目级RAQ评分态度不同的证据。具体来说,DS参与者的决策者对帮助他人的责任问题做出更有利的回应的可能性增加(DS与非DS:赔率比[OR]=1.26,95%CI:1.08,1.48),并且对医学研究将在其一生中找到主要疾病的治愈方法的问题做出更有利的回应的几率降低(DS与非DS:OR=0.77,95%CI:0.66,0.90)。
结论:我们的研究结果为研究人员制定招募有和没有DS的个体进入临床研究的策略提供了见解。观察到的项目级别差异需要进一步调查,以指导准确的招聘策略。
■观察到唐氏综合症(DS)患者的决策者与没有DS的决策者之间的研究态度平均相似。观察到DS和非DS决策者在研究态度上的项目级别差异不同。这些结果可以帮助促进DS人群的精确招募策略。
BACKGROUND: Recruitment challenges in people with and without Down syndrome (DS) can delay research progress and risk sample bias. This study identified and quantified differences in research attitudes across populations of research enrollment decision-makers for individuals with and without DS.
METHODS: We performed analyses using data from two registries: the University of California, Irvine Consent-to-Contact (C2C) Registry and DS-Connect. The former represented a sample of non-DS decision-makers (N = 4818), while for the latter, we excluded individuals with DS, leaving a population of DS family decision-makers (N = 976). We assessed scores on the Research Attitudes Questionnaire (RAQ) between DS and non-DS decision-makers. We compared total RAQ scores using linear regression and assessed item-level RAQ differences using proportional odds regression.
RESULTS: Mean total RAQ scores were not statistically different between decision-makers in the two registries, after adjusting for age, sex, race and ethnicity, education, and the coronavirus disease 2019 (COVID-19) time frame (Est. Diff = 0.11, 95% confidence interval [CI]: -0.22, 0.43; p = 0.531). However, in a pre-specified analysis, we did find evidence of differential attitudes on item-level RAQ scores. Specifically, decision-makers for participants with DS had increased odds of a more favorable response to the question of responsibility to help others (DS vs. non-DS: odds ratio [OR] = 1.26, 95% CI: 1.08, 1.48) and decreased odds of a more favorable response to the question regarding the belief that medical research would find cures for major diseases during their lifetime (DS vs. non-DS: OR = 0.77, 95% CI: 0.66, 0.90).
CONCLUSIONS: Our findings provide insights for researchers to develop strategies for recruiting individuals with and without DS into clinical research. The observed item-level differences warrant further investigation to instruct precise recruitment strategies.
UNASSIGNED: Research attitudes between decision-makers for individuals with Down syndrome (DS) and decision-makers without DS were observed to be similar on average.Item-level differences in research attitudes were observed to differ for DS and non-DS decision-makers.These results can help facilitate precise recruitment strategies for populations with DS.