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Elevidys Clinical Development: Confidence In Surrogate Endpoint A Longstanding Concern For FDA Reviewers

Executive Summary

Timeline of the gene therapy’s clinical development and regulatory review shows agency staff repeatedly questioned the ability of micro-dystrophin to predict clinical benefit in Duchenne muscular dystrophy patients.

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Gene And Cell Therapy Clinical Holds Decline While Those For Drugs Hit 12-Year High

FDA’s Center for Biologics Evaluation and Research saw IND clinical holds for gene and cell therapies dropped to 70 in calendar year 2022, a 52% decline from 2018. Center for Drug Evaluation and Research’s total clinical holds in fiscal year 2022 were 380, the highest number in the past 12 years.

Let’s Go To The Video: Recordings Of DMD Patients After Elevidys Treatment ‘Compelling’ But Not Substantial Evidence

CBER Director Peter Marks urged the review teams for Sarepta’s gene therapy to consider pre- and post-treatment videos showing improved physical function, but clinical and statistical reviewers cited limitations in the patient videos and said they failed to inform whether micro-dystrophin is a suitable surrogate endpoint for accelerated approval.

Sarepta’s External Controls Analysis Weakened By Elevidys Placebo Data Comparison

When placebo data from the only randomized trial of the Duchenne muscular dystrophy gene therapy were compared with Sarepta’s proffered external control data, the placebo subjects numerically outperformed the external controls, ‘calling into question the comparability of the external control group,’ an FDA statistical reviewer said.

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