TY - JOUR
T1 - Doubly-robust methods for differences in restricted mean lifetimes using pseudo-observations
AU - Choi, Sangbum
AU - Choi, Taehwa
AU - Lee, Hye Young
AU - Han, Sung Won
AU - Bandyopadhyay, Dipankar
N1 - Funding Information:
The authors express their gratitude to the three anonymous reviewers, whose constructive comments led to a significantly improved version of the manuscript. The research of S. Choi was supported by grants (2019R1F1A1052239, 2019R1A4A1028, 2022R1A2C1008514) awarded by the National Research Foundation of Korea of the Govt. of South Korea. The research of S. Han was supported by grant (2019R1F1A1060250) awarded by the National Research Foundation of Korea of the Govt. of South Korea. Bandyopadhyay acknowledges partial support from grants (R01DE024984, P30CA016059, P20CA252717 and P20CA264067) awarded by the United States National Institutes of Health.
Funding Information:
National Institute of Dental and Craniofacial Research, Grant/Award Number: R01DE024984; National Institutes of Health, Grant/Award Numbers: P20CA264067, P20CA252717, P30CA016059; National Research Foundation of Korea, Grant/Award Numbers: 2019R1F1A1060250, 2022R1A2C1008514, 2019R1A4A1028, 2019R1F1A1052239 Funding information
Publisher Copyright:
© 2022 John Wiley & Sons Ltd.
PY - 2022/11/1
Y1 - 2022/11/1
N2 - In clinical studies or trials comparing survival times between two treatment groups, the restricted mean lifetime (RML), defined as the expectation of the survival from time 0 to a prespecified time-point, is often the quantity of interest that is readily interpretable to clinicians without any modeling restrictions. It is well known that if the treatments are not randomized (as in observational studies), covariate adjustment is necessary to account for treatment imbalances due to confounding factors. In this article, we propose a simple doubly-robust pseudo-value approach to effectively estimate the difference in the RML between two groups (akin to a metric for estimating average causal effects), while accounting for confounders. The proposed method combines two general approaches: (a) group-specific regression models for the time-to-event and covariate information, and (b) inverse probability of treatment assignment weights, where the RMLs are replaced by the corresponding pseudo-observations for survival outcomes, thereby mitigating the estimation complexities in presence of censoring. The proposed estimator is double-robust, in the sense that it is consistent if at least one of the two working models remains correct. In addition, we explore the potential of available machine learning algorithms in causal inference to reduce possible bias of the causal estimates in presence of a complex association between the survival outcome and covariates. We conduct extensive simulation studies to assess the finite-sample performance of the pseudo-value causal effect estimators. Furthermore, we illustrate our methodology via application to a dataset from a breast cancer cohort study. The proposed method is implementable using the R package drRML, available in GitHub.
AB - In clinical studies or trials comparing survival times between two treatment groups, the restricted mean lifetime (RML), defined as the expectation of the survival from time 0 to a prespecified time-point, is often the quantity of interest that is readily interpretable to clinicians without any modeling restrictions. It is well known that if the treatments are not randomized (as in observational studies), covariate adjustment is necessary to account for treatment imbalances due to confounding factors. In this article, we propose a simple doubly-robust pseudo-value approach to effectively estimate the difference in the RML between two groups (akin to a metric for estimating average causal effects), while accounting for confounders. The proposed method combines two general approaches: (a) group-specific regression models for the time-to-event and covariate information, and (b) inverse probability of treatment assignment weights, where the RMLs are replaced by the corresponding pseudo-observations for survival outcomes, thereby mitigating the estimation complexities in presence of censoring. The proposed estimator is double-robust, in the sense that it is consistent if at least one of the two working models remains correct. In addition, we explore the potential of available machine learning algorithms in causal inference to reduce possible bias of the causal estimates in presence of a complex association between the survival outcome and covariates. We conduct extensive simulation studies to assess the finite-sample performance of the pseudo-value causal effect estimators. Furthermore, we illustrate our methodology via application to a dataset from a breast cancer cohort study. The proposed method is implementable using the R package drRML, available in GitHub.
KW - causal treatment effect
KW - double-robust estimation
KW - inverse probability weighting
KW - pseudo observations
KW - survival analysis
UR - http://www.scopus.com/inward/record.url?scp=85129926405&partnerID=8YFLogxK
U2 - 10.1002/pst.2223
DO - 10.1002/pst.2223
M3 - Article
C2 - 35524651
AN - SCOPUS:85129926405
SN - 1539-1604
VL - 21
SP - 1185
EP - 1198
JO - Pharmaceutical Statistics
JF - Pharmaceutical Statistics
IS - 6
ER -