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Surrogacy Validation for Time-to-Event Outcomes with Illness-Death Frailty Models
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Surrogacy Validation for Time-to-Event Outcomes with Illness-Death Frailty Models

Emily K Roberts, Michael R Elliott and Jeremy M. G Taylor
ArXiv.org
11/28/2022
DOI: 10.48550/arxiv.2211.15826
url
https://doi.org/10.48550/arXiv.2211.15826View
Preprint (Author's original)This preprint has not been evaluated by subject experts through peer review. Preprints may undergo extensive changes and/or become peer-reviewed journal articles. Open Access

Abstract

A common practice in clinical trials is to evaluate a treatment effect on an intermediate endpoint when the true outcome of interest would be difficult or costly to measure. We consider how to validate intermediate endpoints in a causally-valid way when the trial outcomes are time-to-event. Using counterfactual outcomes, those that would be observed if the counterfactual treatment had been given, the causal association paradigm assesses the relationship of the treatment effect on the surrogate $S$ with the treatment effect on the true endpoint $T$. In particular, we propose illness death models to accommodate the censored and semi-competing risk structure of survival data. The proposed causal version of these models involves estimable and counterfactual frailty terms. Via these multi-state models, we characterize what a valid surrogate would look like using a causal effect predictiveness plot. We evaluate the estimation properties of a Bayesian method using Markov Chain Monte Carlo and assess the sensitivity of our model assumptions. Our motivating data source is a localized prostate cancer clinical trial where the two survival endpoints are time to distant metastasis and time to death.
Statistics - Methodology

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