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FDA Approves BioMarin's Gene Therapy for Severe Hemophilia A

NEW YORK – The US Food and Drug Administration on Thursday approved BioMarin's Roctavian (valoctocogene roxaparvovec), making it the first gene therapy for adults with severe hemophilia A.

The FDA had been expected to issue its decision on San Rafael, California-based BioMarin's biologics license application on Friday, an extension from a previously set decision date of March 31. This is the second time that BioMarin has brought the drug before the agency, which rejected the firm's gene therapy application in 2020 and recommended it collect more data from an ongoing Phase III study.

The gene therapy was approved by the European Commission in 2022.

Roctavian is a single-dose gene therapy that uses an adeno-associated virus (AAV) to deliver a functional copy of the FVIII gene that encodes the eponymous protein responsible for blood clotting. Patients with hemophilia A, a condition caused by FVIII gene mutations, have a deficiency of the protein that can lead to uncontrolled bleeding episodes. The treatment is indicated for patients without antibodies to AAV serotype 5, as determined by an FDA-approved test.

The FDA on Thursday also approved a companion diagnostic immunoassay from Salt Lake City-based ARUP Laboratories to detect the presence of preexisting anti-AAV5 antibodies. The AAV5 DetectCDx test, developed in collaboration with BioMarin, is the first companion diagnostic immunoassay approved by the FDA for a gene therapy, according to ARUP. ARUP will serve as the sole laboratory testing provider for Roctavian in the US.

The agency approved Roctavian based on results from a Phase III study, GENEr8-1, comparing patients' annualized bleeding rate (ABR) before and after gene therapy administration. Patients experienced a mean ABR reduction of 52 percent after receiving Roctavian compared to baseline, which was established by collecting ABR for at least six months before receiving the gene therapy, during which they received FVIII prophylaxis as standard care. Patients' baseline ABR was 5.4 bleeds per year compared to 2.6 bleeds per year after the therapy.

The most common adverse reactions to Roctavian were nausea, fatigue, headache, infusion-related reactions, vomiting, and abdominal pain. The therapy is contraindicated for patients with active infections, known significant hepatic fibrosis at stage 3 or 4 or cirrhosis, and a known hypersensitivity to mannitol.

BioMarin said it will continue to monitor long-term effects of Roctavian through an extension study and post-approval studies that will run for up to 15 years.

BioMarin launched Roctavian in Europe with a list price of around €1.5 million ($1.6 million). In the US, the Institute for Clinical and Economic Review, a group that gauges the cost-effectiveness of medical interventions, has estimated one-time Roctavian's Health Benefit Price Benchmark at $1.9 million.

BioMarin in an emailed statement said Roctavian is priced at $2.9 million.

"We set a price that both captures the value of Roctavian and results in material savings to the healthcare system," the company said in the statement. It said FVIII prophylaxis, by contrast, can cost the typical patient about $800,000 per year.

BioMarin said it will offer an outcomes-based warranty to US insurers, in which it will reimburse government and commercial payors for up to 100 percent of the wholesale acquisition cost if a patient receiving Roctavian does not respond to the therapy. If the patient stops responding within the first four years of administering the drug, BioMarin will reimburse the cost on a prorated basis.