Mehrotra Devan V, Marceau West Rachel
Biostatistics and Research Decision Sciences, Merck & Co., Inc., North Wales, Pennsylvania, USA.
Stat Med. 2020 Dec 30;39(30):4724-4744. doi: 10.1002/sim.8750. Epub 2020 Sep 20.
Randomized clinical trials are often designed to assess whether a test treatment prolongs survival relative to a control treatment. Increased patient heterogeneity, while desirable for generalizability of results, can weaken the ability of common statistical approaches to detect treatment differences, potentially hampering the regulatory approval of safe and efficacious therapies. A novel solution to this problem is proposed. A list of baseline covariates that have the potential to be prognostic for survival under either treatment is pre-specified in the analysis plan. At the analysis stage, using all observed survival times but blinded to patient-level treatment assignment, "noise" covariates are removed with elastic net Cox regression. The shortened covariate list is used by a conditional inference tree algorithm to segment the heterogeneous trial population into subpopulations of prognostically homogeneous patients (risk strata). After patient-level treatment unblinding, a treatment comparison is done within each formed risk stratum and stratum-level results are combined for overall statistical inference. The impressive power-boosting performance of our proposed 5-step stratified testing and amalgamation routine (5-STAR), relative to that of the logrank test and other common approaches that do not leverage inherently structured patient heterogeneity, is illustrated using a hypothetical and two real datasets along with simulation results. Furthermore, the importance of reporting stratum-level comparative treatment effects (time ratios from accelerated failure time model fits in conjunction with model averaging and, as needed, hazard ratios from Cox proportional hazard model fits) is highlighted as a potential enabler of personalized medicine. An R package is available at https://github.com/rmarceauwest/fiveSTAR.
随机临床试验通常旨在评估一种试验性治疗相对于对照治疗是否能延长生存期。患者异质性增加虽然有利于结果的普遍性,但会削弱常用统计方法检测治疗差异的能力,可能会阻碍安全有效的疗法获得监管批准。本文提出了一种解决此问题的新方法。在分析计划中预先指定一份基线协变量清单,这些协变量有可能对任何一种治疗下的生存期具有预后价值。在分析阶段,使用所有观察到的生存时间,但对患者层面的治疗分配保持盲态,通过弹性网Cox回归去除“噪声”协变量。条件推断树算法使用缩短后的协变量清单将异质性试验人群划分为预后同质患者的亚组(风险分层)。在患者层面的治疗解盲后,在每个形成的风险分层内进行治疗比较,并将分层层面的结果合并用于总体统计推断。使用一个假设数据集、两个真实数据集以及模拟结果,说明了我们提出的五步分层检验与合并程序(5-STAR)相对于对数秩检验和其他未利用内在结构化患者异质性的常用方法所具有的令人印象深刻的增强检验效能的性能。此外,强调了报告分层层面的比较治疗效果(加速失效时间模型拟合的时间比结合模型平均,并根据需要报告Cox比例风险模型拟合的风险比)作为个性化医疗潜在促成因素的重要性。可在https://github.com/rmarceauwest/fiveSTAR获取一个R包。