Department of Statistics and Actuarial Science, Simon Fraser University, Burnaby, British Columbia, Canada.
Arthritis Research Canada, Vancouver, British Columbia, Canada.
Stat Med. 2022 Jun 15;41(13):2448-2465. doi: 10.1002/sim.9364. Epub 2022 Mar 10.
Treatment noncompliance often occurs in longitudinal randomized controlled trials (RCTs) on human subjects, and can greatly complicate treatment effect assessment. The complier average causal effect (CACE) informs the intervention efficacy for the subpopulation who would comply regardless of assigned treatment and has been considered as patient-oriented treatment effects of interest in the presence of noncompliance. Real-world RCTs evaluating multifaceted interventions often employ multiple study endpoints to measure treatment success. In such trials, limited sample sizes, low compliance rates, and small to moderate effect sizes on individual endpoints can significantly reduce the power to detect CACE when these correlated endpoints are analyzed separately. To overcome the challenge, we develop a multivariate longitudinal potential outcome model with stratification on latent compliance types to efficiently assess multivariate CACEs (MCACE) by combining information across multiple endpoints and visits. Evaluation using simulation data shows a significant increase in the estimation efficiency with the MCACE model, including up to 50% reduction in standard errors (SEs) of CACE estimates and 1-fold increase in the power to detect CACE. Finally, we apply the proposed MCACE model to an RCT on Arthritis Health Journal online tool. Results show that the MCACE analysis detects significant and beneficial intervention effects on two of the six endpoints while estimating CACEs for these endpoints separately fail to detect treatment effect on any endpoint.
在针对人体受试者的纵向随机对照试验(RCT)中,经常会出现治疗不依从的情况,这会极大地增加治疗效果评估的难度。遵从平均因果效应(CACE)为无论分配何种治疗都会遵从的亚组人群提供了干预效果信息,并且在存在不依从的情况下被认为是面向患者的治疗效果。评估多方面干预措施的真实世界 RCT 通常使用多个研究终点来衡量治疗成功。在这些试验中,有限的样本量、低依从率以及对个体终点的小到中等效应大小,在分别分析这些相关终点时,会显著降低检测 CACE 的功效。为了克服这一挑战,我们开发了一种具有潜在依从类型分层的多变量纵向潜在结果模型,通过整合多个终点和访视的信息,有效地评估多变量 CACE(MCACE)。使用模拟数据进行的评估表明,MCACE 模型的估计效率显著提高,包括 CACE 估计值的标准误差(SE)降低多达 50%,以及检测 CACE 的功效提高 1 倍。最后,我们将所提出的 MCACE 模型应用于关节炎健康杂志在线工具的 RCT。结果表明,MCACE 分析在六个终点中的两个终点上检测到了显著且有益的干预效果,而分别对这些终点进行 CACE 分析则未能检测到任何终点的治疗效果。