Department of Statistics, Florida State University, Tallahassee, Florida, USA.
Department of Biostatistics and Computational Biology, University of Rochester, Rochester, New York, USA.
Stat Med. 2023 Jul 10;42(15):2661-2691. doi: 10.1002/sim.9742. Epub 2023 Apr 10.
Existing methods for estimating the mean outcome under a given sequential treatment rule often rely on intention-to-treat analyses, which estimate the effect of following a certain treatment rule regardless of compliance behavior of patients. There are two major concerns with intention-to-treat analyses: (1) the estimated effects are often biased toward the null effect; (2) the results are not generalizable and reproducible due to the potentially differential compliance behavior. These are particularly problematic in settings with a high level of non-compliance, such as substance use disorder studies. Our work is motivated by the Adaptive Treatment for Alcohol and Cocaine Dependence study (ENGAGE), which is a multi-stage trial that aimed to construct optimal treatment strategies to engage patients in therapy. Due to the relatively low level of compliance in this trial, intention-to-treat analyses essentially estimate the effect of being randomized to a certain treatment, instead of the actual effect of the treatment. We obviate this challenge by defining the target parameter as the mean outcome under a dynamic treatment regime conditional on a potential compliance stratum. We propose a flexible non-parametric Bayesian approach based on principal stratification, which consists of a Gaussian copula model for the joint distribution of the potential compliances, and a Dirichlet process mixture model for the treatment sequence specific outcomes. We conduct extensive simulation studies which highlight the utility of our approach in the context of multi-stage randomized trials. We show robustness of our estimator to non-linear and non-Gaussian settings as well.
现有的用于估计给定序贯治疗规则下平均结果的方法通常依赖于意向治疗分析,该分析估计的是遵循特定治疗规则的效果,而不考虑患者的依从行为。意向治疗分析存在两个主要问题:(1)估计效果往往偏向于零效应;(2)由于患者潜在的不同依从行为,结果不可推广和再现。这些问题在非依从性水平较高的情况下(如物质使用障碍研究)尤其突出。我们的工作受到适应性酒精和可卡因依赖治疗研究(ENGAGE)的启发,这是一项多阶段试验,旨在构建最佳治疗策略以吸引患者接受治疗。由于该试验的依从性相对较低,意向治疗分析本质上估计的是被随机分配到特定治疗的效果,而不是治疗的实际效果。我们通过将目标参数定义为潜在依从性分层条件下动态治疗方案的平均结果来规避这一挑战。我们提出了一种基于主分层的灵活非参数贝叶斯方法,其中包括一个潜在依从性联合分布的高斯 Copula 模型,以及一个特定于治疗顺序的结果的狄利克雷过程混合模型。我们进行了广泛的模拟研究,突出了我们的方法在多阶段随机试验中的效用。我们还证明了我们的估计器在非线性和非高斯环境下的稳健性。