Addressing challenges with Matching-Adjusted Indirect Comparisons to demonstrate the comparative effectiveness of entrectinib in metastatic ROS-1 positive Non-Small Cell Lung Cancer

Abstract Background Matching Adjusted Indirect Comparison (MAIC) is a statistical method used to adjust for potential biases when comparing treatment effects between separate data sources, with aggregate data in one arm, and individual patients data in the other. However, acceptance of MAIC in healt...

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Main Authors: Cyril Esnault, Louise Baschet, Vanessa Barbet, Gaëlle Chenuc, Maurice Pérol, Katia Thokagevistk, David Pau, Matthias Monnereau, Lise Bosquet, Thomas Filleron
Format: Article
Language:English
Published: BMC 2025-03-01
Series:BMC Medical Research Methodology
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Online Access:https://doi.org/10.1186/s12874-025-02500-w
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author Cyril Esnault
Louise Baschet
Vanessa Barbet
Gaëlle Chenuc
Maurice Pérol
Katia Thokagevistk
David Pau
Matthias Monnereau
Lise Bosquet
Thomas Filleron
author_facet Cyril Esnault
Louise Baschet
Vanessa Barbet
Gaëlle Chenuc
Maurice Pérol
Katia Thokagevistk
David Pau
Matthias Monnereau
Lise Bosquet
Thomas Filleron
author_sort Cyril Esnault
collection DOAJ
description Abstract Background Matching Adjusted Indirect Comparison (MAIC) is a statistical method used to adjust for potential biases when comparing treatment effects between separate data sources, with aggregate data in one arm, and individual patients data in the other. However, acceptance of MAIC in health technology assessment (HTA) is challenging because of the numerous biases that can affect the estimates of treatment effects – especially with small sample sizes, increasing the risk of convergence issues. We suggest statistical approaches to address some of the challenges in supporting evidence from MAICs, applied to a case study. Methods The proposed approaches were illustrated with a case study comparing an integrated analysis of three single-arm trials of entrectinib with the French standard of care using the Epidemio-Strategy and Medical Economics (ESME) Lung Cancer Data Platform, in metastatic ROS1-positive Non-Small Cell Lung Cancer (NSCLC) patients. To obtain convergent models with balanced treatment arms, a transparent predefined workflow for variable selection in the propensity score model, with multiple imputation of missing data, was used. To assess robustness, multiple sensitivity analyses were conducted, including Quantitative Bias Analyses (QBA) for unmeasured confounders (E-value, bias plot), and for missing at random assumption (tipping-point analysis). Results The proposed workflow was successful in generating satisfactory models for all sub-populations, that is, without convergence problems and with effectively balanced key covariates between treatment arms. It also gave an indication of the number of models tested. Sensitivity analyses confirmed the robustness of the results, including to unmeasured confounders. The QBA performed on the missing data allowed to exclude the potential impact of the missing data on the estimate of comparative effectiveness, even though approximately half of the ECOG Performance Status data were missing. Conclusions To the best of our knowledge, we present the first in-depth application of QBA in the context of MAIC. Despite the real-world data limitations, with this MAIC, we show that it is possible to confirm the robustness of the results by using appropriate statistical methods. Trial registration NA.
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spelling doaj-art-b014117ef5c742afa607db950e4146012025-08-20T02:16:55ZengBMCBMC Medical Research Methodology1471-22882025-03-0125111010.1186/s12874-025-02500-wAddressing challenges with Matching-Adjusted Indirect Comparisons to demonstrate the comparative effectiveness of entrectinib in metastatic ROS-1 positive Non-Small Cell Lung CancerCyril Esnault0Louise Baschet1Vanessa Barbet2Gaëlle Chenuc3Maurice Pérol4Katia Thokagevistk5David Pau6Matthias Monnereau7Lise Bosquet8Thomas Filleron9Roche SASHorianaHorianaBiometry Department, IQVIAMedical Oncology Department, Centre Léon BérardRoche SASRoche SASHorianaHealth Data and Partnership DepartmentBiostatistics & Health Data Science Unit, Institut Claudius Régaud IUCT-OAbstract Background Matching Adjusted Indirect Comparison (MAIC) is a statistical method used to adjust for potential biases when comparing treatment effects between separate data sources, with aggregate data in one arm, and individual patients data in the other. However, acceptance of MAIC in health technology assessment (HTA) is challenging because of the numerous biases that can affect the estimates of treatment effects – especially with small sample sizes, increasing the risk of convergence issues. We suggest statistical approaches to address some of the challenges in supporting evidence from MAICs, applied to a case study. Methods The proposed approaches were illustrated with a case study comparing an integrated analysis of three single-arm trials of entrectinib with the French standard of care using the Epidemio-Strategy and Medical Economics (ESME) Lung Cancer Data Platform, in metastatic ROS1-positive Non-Small Cell Lung Cancer (NSCLC) patients. To obtain convergent models with balanced treatment arms, a transparent predefined workflow for variable selection in the propensity score model, with multiple imputation of missing data, was used. To assess robustness, multiple sensitivity analyses were conducted, including Quantitative Bias Analyses (QBA) for unmeasured confounders (E-value, bias plot), and for missing at random assumption (tipping-point analysis). Results The proposed workflow was successful in generating satisfactory models for all sub-populations, that is, without convergence problems and with effectively balanced key covariates between treatment arms. It also gave an indication of the number of models tested. Sensitivity analyses confirmed the robustness of the results, including to unmeasured confounders. The QBA performed on the missing data allowed to exclude the potential impact of the missing data on the estimate of comparative effectiveness, even though approximately half of the ECOG Performance Status data were missing. Conclusions To the best of our knowledge, we present the first in-depth application of QBA in the context of MAIC. Despite the real-world data limitations, with this MAIC, we show that it is possible to confirm the robustness of the results by using appropriate statistical methods. Trial registration NA.https://doi.org/10.1186/s12874-025-02500-wMAICQBAMissing dataE-valueResidual confounding
spellingShingle Cyril Esnault
Louise Baschet
Vanessa Barbet
Gaëlle Chenuc
Maurice Pérol
Katia Thokagevistk
David Pau
Matthias Monnereau
Lise Bosquet
Thomas Filleron
Addressing challenges with Matching-Adjusted Indirect Comparisons to demonstrate the comparative effectiveness of entrectinib in metastatic ROS-1 positive Non-Small Cell Lung Cancer
BMC Medical Research Methodology
MAIC
QBA
Missing data
E-value
Residual confounding
title Addressing challenges with Matching-Adjusted Indirect Comparisons to demonstrate the comparative effectiveness of entrectinib in metastatic ROS-1 positive Non-Small Cell Lung Cancer
title_full Addressing challenges with Matching-Adjusted Indirect Comparisons to demonstrate the comparative effectiveness of entrectinib in metastatic ROS-1 positive Non-Small Cell Lung Cancer
title_fullStr Addressing challenges with Matching-Adjusted Indirect Comparisons to demonstrate the comparative effectiveness of entrectinib in metastatic ROS-1 positive Non-Small Cell Lung Cancer
title_full_unstemmed Addressing challenges with Matching-Adjusted Indirect Comparisons to demonstrate the comparative effectiveness of entrectinib in metastatic ROS-1 positive Non-Small Cell Lung Cancer
title_short Addressing challenges with Matching-Adjusted Indirect Comparisons to demonstrate the comparative effectiveness of entrectinib in metastatic ROS-1 positive Non-Small Cell Lung Cancer
title_sort addressing challenges with matching adjusted indirect comparisons to demonstrate the comparative effectiveness of entrectinib in metastatic ros 1 positive non small cell lung cancer
topic MAIC
QBA
Missing data
E-value
Residual confounding
url https://doi.org/10.1186/s12874-025-02500-w
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