Wey Andrew, Vock David M, Connett John, Rudser Kyle
Minneapolis Medical Research Foundation, Minneapolis, MN, U.S.A.
Biostatistics and Data Management Core, John A. Burns School of Medicine, University of Hawaii, Honolulu, Hawaii.
Stat Med. 2016 Aug 30;35(19):3319-32. doi: 10.1002/sim.6929. Epub 2016 Mar 2.
The difference in restricted mean survival times between two groups is a clinically relevant summary measure. With observational data, there may be imbalances in confounding variables between the two groups. One approach to account for such imbalances is estimating a covariate-adjusted restricted mean difference by modeling the covariate-adjusted survival distribution and then marginalizing over the covariate distribution. Because the estimator for the restricted mean difference is defined by the estimator for the covariate-adjusted survival distribution, it is natural to expect that a better estimator of the covariate-adjusted survival distribution is associated with a better estimator of the restricted mean difference. We therefore propose estimating restricted mean differences with stacked survival models. Stacked survival models estimate a weighted average of several survival models by minimizing predicted error. By including a range of parametric, semi-parametric, and non-parametric models, stacked survival models can robustly estimate a covariate-adjusted survival distribution and, therefore, the restricted mean treatment effect in a wide range of scenarios. We demonstrate through a simulation study that better performance of the covariate-adjusted survival distribution often leads to better mean squared error of the restricted mean difference although there are notable exceptions. In addition, we demonstrate that the proposed estimator can perform nearly as well as Cox regression when the proportional hazards assumption is satisfied and significantly better when proportional hazards is violated. Finally, the proposed estimator is illustrated with data from the United Network for Organ Sharing to evaluate post-lung transplant survival between large-volume and small-volume centers. Copyright © 2016 John Wiley & Sons, Ltd.
两组受限平均生存时间的差异是一种具有临床相关性的汇总指标。对于观察性数据,两组之间的混杂变量可能存在不均衡。处理此类不均衡的一种方法是,通过对协变量调整后的生存分布进行建模,然后对协变量分布进行边际化,来估计协变量调整后的受限平均差异。由于受限平均差异的估计量是由协变量调整后的生存分布的估计量定义的,因此可以自然地预期,协变量调整后的生存分布的更好估计量与受限平均差异的更好估计量相关。因此,我们建议使用堆叠生存模型来估计受限平均差异。堆叠生存模型通过最小化预测误差来估计几个生存模型的加权平均值。通过纳入一系列参数模型、半参数模型和非参数模型,堆叠生存模型能够在广泛的情形中稳健地估计协变量调整后的生存分布,进而估计受限平均治疗效果。我们通过一项模拟研究表明,尽管存在显著例外情况,但协变量调整后的生存分布的更好性能通常会导致受限平均差异的均方误差更小。此外,我们证明,当满足比例风险假设时,所提出的估计量的性能几乎与Cox回归一样好,而当比例风险假设不成立时,其性能则显著更好。最后,我们用器官共享联合网络的数据说明了所提出的估计量,以评估大容量中心和小容量中心之间肺移植后的生存情况。版权所有© 2016约翰威立父子有限公司。