Restricted mean survival time in cluster randomized trials with a small number of clusters: Improving variance estimation of the intervention effect from the pseudo-values regression

F. Le Vilain-Abraham et al.

Statistical Methods In Medical Research2026https://doi.org/10.1177/09622802251406581article
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What the paper says

In randomized clinical trials with a time-to-event outcome, the intervention effect could be quantified by a difference in restricted mean survival time (ΔRMST) between the intervention and control groups, defined as the expected survival duration gain due to the intervention over a fixed follow-up period. In cluster randomized trials (CRTs), social units are randomized to intervention or control groups; the correlation between survival times of the individuals within the same cluster must be taken into account in the statistical analysis. In a previous work, we proposed the use of pseudo-values regression, based on generalized estimating equations (GEEs), for estimating ΔRMST in CRTs. We showed that this method correctly estimated the ΔRMST and controlled the type I error rate in CRTs with at least 50 clusters. Here, we propose methods for CRTs with a small number of clusters (<50). We evaluated the performance of four bias-corrections of the GEE sandwich variance estimator of the intervention effect. We also considered the use of a Student <i>t</i> distribution as an alternative to the normal distribution of the GEE Wald test statistic for testing the intervention effect and constructing the confidence interval. With a simulation study, assuming proportional or non-proportional hazards, we showed that the Student <i>t</i> distribution outperformed the normal distribution in terms of type I error rate, and the Fay and Graubard bias-corrected variance led to an appropriate type I error rate whatever the number of clusters. Therefore, we recommend the use of the Fay and Graubard variance estimator combined with a Student <i>t</i> distribution for the pseudo-values regression to correctly estimate the variance of the intervention effect. Finally, we provide an illustrative analysis of the DEMETER trial evaluating the use of a specific endotracheal tube for subglottic secretion drainage to prevent ventilator-associated pneumonia, by comparing each of the methods considered.

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https://doi.org/https://doi.org/10.1177/09622802251406581

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@article{f.2026,
  title        = {{Restricted mean survival time in cluster randomized trials with a small number of clusters: Improving variance estimation of the intervention effect from the pseudo-values regression}},
  author       = {F. Le Vilain-Abraham et al.},
  journal      = {Statistical Methods In Medical Research},
  year         = {2026},
  doi          = {https://doi.org/https://doi.org/10.1177/09622802251406581},
}

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Evidence weight

0.50

Balanced mode · F 0.40 / M 0.15 / V 0.05 / R 0.40

F · citation impact0.50 × 0.4 = 0.20
M · momentum0.50 × 0.15 = 0.07
V · venue signal0.50 × 0.05 = 0.03
R · text relevance †0.50 × 0.4 = 0.20

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