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Surrogacy of Pathologic Complete Response in Trials of Neoadjuvant Therapy for Early Breast CancerCritical Analysis of Strengths, Weaknesses, and Misinterpretations

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Abstract

Importance  The pathologic complete response (pCR) is supported by regulatory agencies as a surrogate end point for long-term patients’ clinical outcomes in the accelerated approval process of new drugs tested in neoadjuvant randomized clinical trials (RCTs) for early breast cancer (BC). However, a meaningful association between pCR and patients’ survival has been proven only at the patient level (ie, significantly better survival of patients who achieved pCR compared with those who did not), but not at trial level (ie, poor association between degree of improvement in pCR rate and survival reported across trials).

Observations  We critically discuss the potential reasons of such discrepancy between pCR surrogacy value at the patient and trial level, as well as the relevant implications for both clinical research and drug regulatory policy. We also describe alternative surrogate end points, including combined end points that jointly analyzed pathological response and event-free survival data, or the assessment of circulating tumor DNA (ctDNA). Such proposed surrogate end points could overcome limits of pCR and provide a reasonable trade-off between the 2 conflicting needs to have access to effective therapies rapidly, and to reliably assess patients’ clinical benefit.

Conclusions and Relevance  Using surrogate end points to grant drug approvals is justified only when they can provide accurate prediction of a drug’s effect on the long-term patient outcomes. Evidence currently available does not support pCR used alone as a reliable surrogate end point in regulatory neoadjuvant RCTs for BC. The surrogacy value at trial level of potentially more robust surrogate end points needs to be urgently tested.

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Article Information

Accepted for Publication: June 7, 2022.

Published Online: October 6, 2022. doi:10.1001/jamaoncol.2022.3755

Corresponding Author: Fabio Conforti, MD, Division of Medical Oncology for Melanoma & Sarcoma, European Institute of Oncology, Via Ripamonti 435, 20141, Milan, Italy (fabio.conforti@ieo.it).

Author Contributions: Dr Conforti had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis. Authors Conforti, Pala, Viale, and Gelber contributed equally to the work.

Concept and design: Conforti, Pala, Bagnardi, De Pas, Colleoni, Buyse, Gianni, Winer, Loibl, Cortés, Viale.

Acquisition, analysis, or interpretation of data: Conforti, Bagnardi, Hortobagyi, Loibl, Piccart-Gebhart, Wolff, Gelber.

Drafting of the manuscript: Conforti, Pala, Bagnardi, De Pas, Colleoni, Buyse.

Critical revision of the manuscript for important intellectual content: Conforti, Pala, Bagnardi, De Pas, Hortobagyi, Gianni, Winer, Loibl, Cortés, Piccart-Gebhart, Wolff, Viale, Gelber.

Statistical analysis: Conforti, Bagnardi.

Administrative, technical, or material support: ConfortI, Gelber.

Supervision: Conforti, Pala, Bagnardi, Gianni, Loibl, Cortés, Piccart-Gebhart, Viale.

Other: Winer.

Conflict of Interest Disclosures: Dr Colleoni reported grants from Roche outside the submitted work. Dr Buyse reported nonfinancial support from IDDI, stockholder outside the submitted work. Dr Hortobagyi reported personal fees from Novartis outside the submitted work. Dr Gianni reported advisory board membership, consultancy fees, or support to institution from AstraZeneca, Celgene, Eli Lilly, Roche, Pfizer, Seattle Genetics, Artemida Pharma Ltd, Zymeworks; from METIS Precision Medecine, Novartis, Revolution Medicines, Synaffix, Menarini Ricerche, Amgen, Biomedical Insights, and Denali Therapeutics, outside the submitted work; in addition, Dr Gianni had a patent for European Patent Application n. 12195182.6 and 12196177.5; Roche; no compensation was provided. Dr Winer reported consultancy fees from Carrick Therapeutics; honoraria from Genentech/Roche; consultancy fees for advisory board membership, lectures or writing fees, or fees paid to institution from Genomic Helath, GSK, Jounce Therapeutics, and Leap Therapeutics outside the submitted work. Dr Loibl reported honoraria for advisory board membership, lecture and/or writing fees, and/or fees paid to institution from Abbvie, Amgen, AstraZeneca, BMS, Celgene, EirGenix, GSK, Gilead, Lilly, Merck, Novartis, Pfizer, Pierre Fabre, Seagen, DSI, Sanofi, Roche, outside the submitted work; in addition, Dr Loibl had a patent for EP14153692.0 pending Immunsignature in TNBC, paid to institute, a patent for EP21152186.9 pending Signature for CDK 4/6 Inhibitor, paid to institute, a patent for EP15702464.7 issued predicting response to an Anti–HER2-containing therapy, paid to institute, a patent for EP19808852.8 pending GeparNuevo, paid to institute, and a patent for Digital Ki67 Evaluator with royalties paid VM Scope GmbH, paid to institute. Dr Cortés reported personal fees, honoraria for advisory board membership, lecture and/or writing fees, travel expenses, reasearch funding, stock holdings, and/or fees paid to institution from Roche, Celgene, Cellestia Consulting, AstraZeneca, Seattle Genetics, Daiichi Sankyo, Erytech, Athenex, Polyphor, Lilly, Merck Sharp & Dohme, GSK, Leuko, Bioasis, Clovis Oncology, Boehringer, Ellipses, Hibercell, Gemoab, Gilead, Menarini, Zymeworks, Reveal Genomics, Novartis, Eisai, Pfizer, Samsung, Roche, Ariad Pharmaceuticals, AstraZeneca, Baxalta, Bayer Healthcare, Guardanth Health, Merck Sharp & Dohme, Piqur Therapeutics, Puma C, Queen Mary University of London, Medsir Nektar Therapeutics, Leuko, during the conduct of the study; personal fees, honoraria for advisory board membership, lecture and/or writing fees, travel expenses, reasearch funding, stock holdings, and/or fees paid to institution Roche, Celgene, Cellestia, AstraZeneca, Seattle Genetics, Daiichi Sankyo, Erytech, Athenex, Polyphor, Lilly, Merck Sharp & Dohme, GSK, Leuko, Bioasis, Clovis Oncology, Boehringer, Ellipses, Hibercell, Gemoab, Gilead, Menarini, Zymeworks, Reveal Genomics, Novartis, Eisai, Pfizer, Samsung Bioepis, Roche, Ariad Pharmaceuticals, AstraZeneca, Baxalta, Bayer Healthcare, Guardanth Health, Merck Sharp & Dohme, Piqur Therapeutics, Puma C, Queen Mary University of London, Medsir, Nektar Therapeutics, and Leuko, outside the submitted work; in addition, Dr Cortés had a patent for Pharmaceutical Combinations of A Pi3k Inhibitor And A Microtubule Destabilizing Agent. Javier Cortés Castán, Alejandro Piris Giménez, Violeta Serra Elizalde issued WO 2014/199294 A and a patent for Her2 as a predictor of response to dual HER2 blockade in the absence of cytotoxic therapy. Aleix Prat, Antonio Llombart, Javier Cortés issued US 2019/0338368 A1. Dr Piccart-Gebhart reported personal fees, honoraria for advisory board membership, lecture and/or writing fees, travel expenses, reasearch funding, stock holdings, and/or fees paid to institution from AstraZeneca, Camel-IDS/Precirix, Frame Therapeutics, Gilead, Immunomedics, Immutep, Lilly, Menarini, MSD, NBE Therapeutics, Novartis, Odonate, Pfizer, Roche-Genentech, SeaGen, Seattle Genetics, Oncolytics, AstraZeneca, Immunomedics, Menarini, Radius, Servier, and Synthon outside the submitted work. Dr Viale reported personal, advisory board, and/or consultancy fees from Roche, AstraZeneca, MSD, Daichii Sankyo, Agilent, Ventana, and Pfizer outside the submitted work. Dr Gelber reported grants from Roche, AstraZeneca, Merck, and Novartis to institution outside the submitted work. No other disclosures were reported.

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