Evidence map›Paper›PMID 40263938›Full record

ReviewMolecular therapy : the journal of the American Society of Gene Therapy2025

Clinical perspective: Advancing hemophilia treatment through gene therapy approaches.

Courtney D Thornburg, Steve W Pipe, Alessio Cantore, Carmen Unzu, Micheala Jones, Wolfgang A Miesbach

Abstract readReview
In one paragraph

Review in Molecular therapy : the journal of the American Society of Gene Therapy, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed
–field-weighted citation impact
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

5 citing papers in PubMed.

  1. Article
  2. Article
  3. Review
  4. Article
  5. Defining the Role of Nurses in Gene Therapy for Haemophilia.Haemophilia : the official journal of the World Federation of Hemophilia
    Article
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.

Courtney D ThornburgNational Institutes of Health, National Heart, Lung, and Blood Institute, Division of Blood Diseases and Resources, Bethesda, MD, USA.
Steve W PipePediatric Hematology-Oncology, University of Michigan, Ann Arbor, MI, USA.
Alessio CantoreSan Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, Milan, Italy; Vita-Salute San Raffaele University, Milan, Italy.
Carmen UnzuDNA and RNA Medicine Division, CIMA Universidad de Navarra, Pamplona, Spain.
Micheala JonesRegeneron Pharmaceuticals, Inc., Tarrytown, NY, USA.
Wolfgang A MiesbachDepartment of Haemostaseology University Hospital Frankfurt, Frankfurt, Germany. Electronic address: miesbach@em.uni-frankfurt.de.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Hemophilia, a congenital bleeding disorder, can cause arthropathy, impaired mobility, pain, and life-threatening hemorrhage events, significantly impacting quality of life for patients and caregivers. Current therapies, although effective, necessitate costly lifelong treatment, often in specialized settings. However, as a monogenic disorder caused by loss-of-function genetic variants, hemophilia is amenable to gene therapy. In this article, three primary gene therapy approaches at the forefront of clinical development are reviewed. Adeno-associated virus-based gene therapy, having secured approval in the EU, UK, and US after promising phase 3 trial results, demonstrates clear superiority over standard-of-care treatment. Lentivirus-based approaches capable of transducing dividing and nondividing cells may improve the durability of treatment and have low susceptibility to pre-existing neutralizing antibodies to viral vectors. Finally, gene editing techniques such as zinc finger nucleases and CRISPR aim to correct genetic defects directly, holding promise as novel, effective, and highly durable therapeutic strategies in adults and children with hemophilia. This review provides a comprehensive summary of the current status of these gene therapy approaches, highlighting advantages, limitations, and potential future developments.

Indexed as

Genetic TherapyHemophilia AAnimalsClinical Trials as TopicCRISPR-Cas SystemsDependovirusGene EditingGenetic VectorsHumansLentivirusadvanced gene therapiesCRISPR-Cas9efficacygene editinghemophiliapediatricsafety

Identifiers

PMID40263938
PMCPMC12172285

What OpenQuestion holds

Textmetadata
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.