Evidence map›Paper›PMID 40790091›Full record

ReviewGene therapy2025

Gene editing for collagen disorders: current advances and future perspectives.

Klaudia Kocsy, Harry Wilkinson, Favour Felix-Ilemhenbhio, Benjamin Bax, Tom Van Agtmael, Mimoun Azzouz, Arshad Majid

Abstract readReview
In one paragraph

Review in Gene therapy, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing 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

4 citing papers in PubMed.

  1. Review
  2. Review
  3. Review
  4. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

7 authors.

Klaudia KocsySchool of Medicine and Population Health, Sheffield Institute for Translational Neuroscience (SITraN), University of Sheffield, Sheffield, UK. k.kocsy@sheffield.ac.uk.ORCID 0000-0002-8007-8937
Harry WilkinsonSchool of Medicine and Population Health, Sheffield Institute for Translational Neuroscience (SITraN), University of Sheffield, Sheffield, UK.ORCID 0009-0001-6949-7380
Favour Felix-IlemhenbhioSchool of Medicine and Population Health, Sheffield Institute for Translational Neuroscience (SITraN), University of Sheffield, Sheffield, UK.
Benjamin BaxMedicines Discovery Institute, Cardiff University, Cardiff, UK.
Tom Van AgtmaelSchool of Cardiovascular & Metabolic Health, University of Glasgow, Glasgow, UK.ORCID 0000-0003-4282-449X
Mimoun Azzouz *School of Medicine and Population Health, Sheffield Institute for Translational Neuroscience (SITraN), University of Sheffield, Sheffield, UK.
Arshad Majid *School of Medicine and Population Health, Sheffield Institute for Translational Neuroscience (SITraN), University of Sheffield, Sheffield, UK.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Collagen disorders encompass a wide range of genetic conditions caused by pathogenic variants in collagen genes for which there is an unmet need for treatments. They present various clinical features, ranging from localised tissue abnormalities to severe systemic complications. Symptoms differ significantly and depend on the pathogenic variant, which can affect various systems, including the musculoskeletal, cardiovascular, and respiratory systems, highlighting the complex implications of collagen gene pathogenic variants and the wide range of expression patterns among different collagen types. Gene-editing technologies, particularly Clustered Regularly Interspaced Palindromic Repeats (CRISPR)-Cas systems, have emerged as promising therapeutic options for these disorders, representing a putative one-for-all treatment strategy. This review provides an overview of current gene-editing strategies aimed at collagen-related diseases, including osteogenesis imperfecta, Alport syndrome, and dystrophic epidermolysis bullosa. We explore the application of CRISPR-Cas9, which facilitates targeted DNA modifications, base editing (BE), and prime editing (PE), enabling precise single-nucleotide alterations without double-strand breaks (DSB). Preclinical and clinical studies have shown the potential of gene therapy to enhance collagen production, restore tissue integrity, and alleviate symptoms. However, challenges persist, including the lack of recurring mutations, the need for improved delivery methods, the reduction of off-target effects, and the development of novel therapies. Despite these challenges, advancements in gene editing techniques appear promising in enhancing editing efficiency while minimising unintended mutations, paving the way for more precise and safer genetic interventions for collagen disorders. Gene editing is fundamentally transforming medicine and biotechnology. Its applications encompass advanced diagnostics, tailored therapeutic strategies, and solutions for rare genetic disorders. By enabling precise genetic modifications, gene editing is paving the way for treatments of previously untreatable diseases, including those linked to collagen pathogenic variants. This review discusses the latest advancements in gene therapy techniques targeting collagen-related disorders. It explores innovative approaches like CRISPR-Cas9-mediated gene editing and highlights emerging strategies, such as allele-specific inactivation and base editing (BE). By examining these cutting-edge therapies and their potential clinical applications, this review highlights the transformative impact of gene editing in treating collagen-related conditions, while also identifying critical challenges and future directions for research.

Indexed as

Collagen DiseasesGene EditingGenetic TherapyAnimalsCollagenCRISPR-Cas SystemsHumansOsteogenesis ImperfectaCollagen

Identifiers

PMID40790091
PMCPMC12714581

What OpenQuestion holds

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Registered trials

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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.