A Reflection on the Impact of Misspecifying Unidentifiable Causal Inference Models in Surrogate Endpoint Evaluation

Fuente: arXiv
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Autores principales: Deliorman, Gokce, Stijven, Florian, Van der Elst, Wim, Pardo, Maria del Carmen, Alonso, Ariel
Formato: Preprint
Publicado: 2024
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author Deliorman, Gokce
Stijven, Florian
Van der Elst, Wim
Pardo, Maria del Carmen
Alonso, Ariel
author_facet Deliorman, Gokce
Stijven, Florian
Van der Elst, Wim
Pardo, Maria del Carmen
Alonso, Ariel
contents Surrogate endpoints are often used in place of expensive, delayed, or rare true endpoints in clinical trials. However, regulatory authorities require thorough evaluation to accept these surrogate endpoints as reliable substitutes. One evaluation approach is the information-theoretic causal inference framework, which quantifies surrogacy using the individual causal association (ICA). Like most causal inference methods, this approach relies on models that are only partially identifiable. For continuous outcomes, a normal model is often used. Based on theoretical elements and a Monte Carlo procedure we studied the impact of model misspecification across two scenarios: 1) the true model is based on a multivariate t-distribution, and 2) the true model is based on a multivariate log-normal distribution. In the first scenario, the misspecification has a negligible impact on the results, while in the second, it has a significant impact when the misspecification is detectable using the observed data. Finally, we analyzed two data sets using the normal model and several D-vine copula models that were indistinguishable from the normal model based on the data at hand. We observed that the results may vary when different models are used.
format Preprint
id arxiv_https___arxiv_org_abs_2410_04438
institution arXiv
publishDate 2024
record_format arxiv
spellingShingle A Reflection on the Impact of Misspecifying Unidentifiable Causal Inference Models in Surrogate Endpoint Evaluation
Deliorman, Gokce
Stijven, Florian
Van der Elst, Wim
Pardo, Maria del Carmen
Alonso, Ariel
Methodology
Statistics Theory
Applications
Surrogate endpoints are often used in place of expensive, delayed, or rare true endpoints in clinical trials. However, regulatory authorities require thorough evaluation to accept these surrogate endpoints as reliable substitutes. One evaluation approach is the information-theoretic causal inference framework, which quantifies surrogacy using the individual causal association (ICA). Like most causal inference methods, this approach relies on models that are only partially identifiable. For continuous outcomes, a normal model is often used. Based on theoretical elements and a Monte Carlo procedure we studied the impact of model misspecification across two scenarios: 1) the true model is based on a multivariate t-distribution, and 2) the true model is based on a multivariate log-normal distribution. In the first scenario, the misspecification has a negligible impact on the results, while in the second, it has a significant impact when the misspecification is detectable using the observed data. Finally, we analyzed two data sets using the normal model and several D-vine copula models that were indistinguishable from the normal model based on the data at hand. We observed that the results may vary when different models are used.
title A Reflection on the Impact of Misspecifying Unidentifiable Causal Inference Models in Surrogate Endpoint Evaluation
topic Methodology
Statistics Theory
Applications
url https://arxiv.org/abs/2410.04438