Evidence map›Paper›PMID 36111754›Full record

ReviewCurrent gene therapy2023

Characteristics of BAY 2599023 in the Current Treatment Landscape of Hemophilia A Gene Therapy.

Steven W Pipe, Valder R Arruda, Claudia Lange, Stephen Kitchen, Hermann Eichler, Samuel Wadsworth

Open access · greenAbstract readReview
In one paragraph

Review in Current gene therapy, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.

0numbers the graph read from it
0cells of the map it votes in
7citing papers in PubMed
1.3field-weighted citation impact, top 18% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

7 citing papers in PubMed, 8 citations in OpenAlex.

  1. Review
  2. Review
  3. Gene therapy for hemophilia - From basic science to first approvals of "one-and-done" therapies.Molecular therapy : the journal of the American Society of Gene Therapy · 2025
    Review
  4. Gene therapy in hemophilia: the dawn of a new era.Research and practice in thrombosis and haemostasis · 2025
    Review
  5. Article
  6. Review
  7. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

6 authors at 6 institutions in 3 countries.

Steven W PipeDepartments of Pediatrics and Pathology, University of Michigan, Ann Arbor, MI, USA.
Valder R ArrudaDivision of Hematology, Department of Pediatrics, Center for Cell and Molecular Therapeutics at Children's Hospital of Philadelphia, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.
Claudia LangeBayer AG, Berlin, Germany.
Stephen KitchenSheffield Haemophilia and Thrombosis Centre, Sheffield Teaching Hospitals, Sheffield, UK.
Hermann EichlerInstitute of Clinical Hemostaseology and Transfusion Medicine, Saarland University and University Hospital, Homburg/Saar, Germany.
Samuel WadsworthUltragenyx Pharmaceutical Inc., Novato, CA, USA.
Bayer (Germany) · DEChildren's Hospital of Philadelphia · USSaarland University · DESheffield Teaching Hospitals NHS Foundation Trust · GBUltragenyx Pharmaceutical (United States) · USUniversity of Michigan · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Hemophilia A, a single gene disorder leading to deficient Factor VIII (FVIII), is a suitable candidate for gene therapy. The aspiration is for single administration of a genetic therapy that would allow the production of endogenous FVIII sufficient to restore hemostasis and other biological processes. This would potentially result in reliable protection from bleeding and its associated physical and emotional impacts. Gene therapy offers the possibility of a clinically relevant improvement in disease phenotype and transformational improvement in quality of life, including an opportunity to engage in physical activities more confidently. Gene therapy products for hemophilia A in advanced clinical development use adeno-associated viral (AAV) vectors and a codon-optimized B-domain deleted FVIII transgene. However, the different AAV-based gene therapies have distinct design features, such as choice of vector capsid, enhancer and promoter regions, FVIII transgene sequence and manufacturing processes. These, in turn, impact patient eligibility, safety and efficacy. Ideally, gene therapy technology for hemophilia A should offer bleed protection, durable FVIII expression, broad eligibility and limited response variability between patients, and long-term safety. However, several limitations and challenges must be overcome. Here, we introduce the characteristics of the BAY 2599023 (AAVhu37.hFVIIIco, DTX 201) gene therapy product, including the low prevalence in the general population of anti-AAV-hu37 antibodies, as well as other gene therapy AAV products and approaches. We will examine how these can potentially meet the challenges of gene therapy, with the ultimate aim of improving the lives of patients with hemophilia A.

Indexed as

Hemophilia AAnimalsDependovirusGenetic TherapyHumansQuality of LifeAAV vectorBAY 2599023factor VIIIgene therapyhemophilia Amanufacturingtreatment landscape

Identifiers

PMID36111754
PMCPMC10186383
OpenAlexW4296025838

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.