Translating questions to estimands in randomized clinical trials with intercurrent events

被引:12
|
作者
Stensrud, Mats J. [1 ]
Dukes, Oliver [2 ,3 ]
机构
[1] Ecole Polytech Fed Lausanne, Dept Math, Lausanne, Switzerland
[2] Univ Penn, Wharton Sch, Dept Stat & Data Sci, Philadelphia, PA 19104 USA
[3] Univ Ghent, Dept Appl Math Stat & Comp Sci, Ghent, Belgium
关键词
causal inference; estimands; identification; intercurrent events; principal stratification; PRINCIPAL STRATIFICATION; EXPOSURE-RESPONSE; CAUSAL INFERENCE; IDENTIFIABILITY; TRASTUZUMAB; MEDIATION; DESIGNS;
D O I
10.1002/sim.9398
中图分类号
Q [生物科学];
学科分类号
07 ; 0710 ; 09 ;
摘要
Intercurrent (post-treatment) events occur frequently in randomized trials, and investigators often express interest in treatment effects that suitably take account of these events. Contrasts that naively condition on intercurrent events do not have a straight-forward causal interpretation, and the practical relevance of other commonly used approaches is debated. In this work, we discuss how to formulate and choose an estimand, beyond the marginal intention-to-treat effect, from the point of view of a decision maker and drug developer. In particular, we argue that careful articulation of a practically useful research question should either reflect decision making at this point in time or future drug development. Indeed, a substantially interesting estimand is simply a formalization of the (plain English) description of a research question. A common feature of estimands that are practically useful is that they correspond to possibly hypothetical but well-defined interventions in identifiable (sub)populations. To illustrate our points, we consider five examples that were recently used to motivate consideration of principal stratum estimands in clinical trials. In all of these examples, we propose alternative causal estimands, such as conditional effects, sequential regime effects, and separable effects, that correspond to explicit research questions of substantial interest.
引用
收藏
页码:3211 / 3228
页数:18
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