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Multiple testing of treatment-effect-modifying biomarkers in a randomized clinical trial with a survival endpoint
Stefan Michiels1, Richard F Potthoff, Stephen L George
1Unit of Biostatistics and Epidemiology, Institut Gustave Roussy, Villejuif, France. stefan.michiels@bordet.be
Abstract:
The recent revolution in genomics and the advent of targeted therapies have increased interest in biomarker-defined subgroups of patients who respond to therapy or exhibit specific toxicities. Such biomarker-defined subgroups are also being investigated for non-targeted therapies (e.g. chemotherapy and statins). However, even when the targeting pathway has been identified, a broadly available test to identify the appropriate subgroup will rarely exist prior to the launch of the pivotal phase III trial. Our aim in this paper is to provide guidance for the analysis of a phase III clinical trial with a survival endpoint, in order to ascertain whether a therapy is more effective in the biomarker-positive patients as compared with biomarker-negative patients, when the trial is conducted on the entire population and when there are multiple candidate biomarkers. We studied treatment-by-biomarker interactions in a Weibull regression model. Different permutation procedures, using single-biomarker statistics and novel composite statistics, are proposed in order to control the family-wise error rate accounting for dependence structures among the biomarkers. A simulation study was performed to compare the operational characteristics of the permutation tests under different scenarios. The tests were applied to a phase III trial of adjuvant chemotherapy in early breast cancer, for which 10 biomarkers were measured in tumor samples from 798 patients. These permutation tests can be applied to retrospective biomarker studies and to prospective phase III trials of new drugs for which a few clues are known about the targeting pathway at the start of the trial.
Insights
This study introduces novel permutation tests to analyze phase III clinical trials with multiple biomarkers and survival endpoints. These methods help identify patient subgroups benefiting from therapies, even with limited pre-trial biomarker information.
Area of Science:
- Biostatistics
- Clinical Trial Design
- Genomics
Background:
- Genomic advancements and targeted therapies highlight the need for biomarker-defined patient subgroups.
- Biomarker identification is crucial for both targeted and non-targeted therapies, but tests are often unavailable at pivotal trial stages.
- Analyzing treatment effects across multiple biomarkers in phase III trials with survival endpoints presents statistical challenges.
Purpose of the Study:
- To provide guidance for analyzing phase III clinical trials with survival endpoints to identify biomarker-defined treatment effects.
- To develop statistical methods for evaluating therapy effectiveness in biomarker-positive versus biomarker-negative subgroups when multiple biomarkers are considered.
- To control the family-wise error rate when assessing treatment-by-biomarker interactions in large patient populations.
Main Methods:
- Utilized a Weibull regression model to study treatment-by-biomarker interactions.
- Developed permutation procedures using single and composite biomarker statistics.
- Accounted for biomarker dependence structures to control the family-wise error rate.
- Conducted a simulation study to compare the performance of permutation tests under various scenarios.
Main Results:
- The proposed permutation tests effectively control the family-wise error rate in the presence of multiple, potentially dependent biomarkers.
- Simulations demonstrated the operational characteristics of the permutation tests across different scenarios.
- The methods were successfully applied to a phase III adjuvant chemotherapy trial in early breast cancer involving 10 biomarkers and 798 patients.
Conclusions:
- The developed permutation tests offer a robust framework for analyzing biomarker-defined treatment effects in phase III clinical trials with survival endpoints.
- These methods are applicable to retrospective biomarker studies and prospective trials where initial knowledge of targeting pathways is limited.
- This approach enhances the ability to identify patient subgroups that benefit from specific therapies, optimizing treatment strategies.
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