Cited by 190Retinal Development and DisordersRetinal Diseases and TreatmentsVirus-based gene therapy research

Early and late stage gene therapy interventions for inherited retinal degenerations

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Botto, Catherine, Rucli, Marco, Tekinsoy, Müge Defne, Pulman, Juliette, Sahel, José-Alain, Dalkara, Deniz·2022·Progress in Retinal and Eye Research

Abstract

Inherited and age-related retinal degeneration is the hallmark of a large group of heterogeneous diseases and is the main cause of untreatable blindness today. Genetic factors play a major pathogenic role in retinal degenerations for both monogenic diseases (such as retinitis pigmentosa) and complex diseases with established genetic risk factors (such as age-related macular degeneration). Progress in genotyping techniques and back of the eye imaging are completing our understanding of these diseases and their manifestations in patient populations suffering from retinal degenerations. It is clear that whatever the genetic cause, the majority of vision loss in retinal diseases results from the loss of photoreceptor function. The timing and circumstances surrounding the loss of photoreceptor function determine the adequate therapeutic approach to use for each patient. Among such approaches, gene therapy is rapidly becoming a therapeutic reality applicable in the clinic. This massive move from laboratory work towards clinical application has been propelled by the advances in our understanding of disease genetics and mechanisms, gene delivery vectors, gene editing systems, and compensatory strategies for loss of photoreceptor function. Here, we provide an overview of existing modalities of retinal gene therapy and their relevance based on the needs of patient populations suffering from inherited retinal degenerations.

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References 183 · Cited by 190

Research works printed but not linked: 1

  1. Audo, I., Manes, G., Mohand-Saïd, S., Friedrich, A., Lancelot, M.-E., Antonio, A., Moskova-Doumanova, V., Poch, O., Zanlonghi, X., Hamel, Christian P., et al., 2010a. Spectrum of rhodopsin mutations in French autosomal dominant rod-cone dystrophy patients. Invest. Opthalmol. Vis. Sci. 51 (7), 3687. https://doi.org/ 10.1167/iovs.09-4766. · DOI

Authors
Botto, C., Rucli, M., Tekinsoy, M. D., Pulman, J., Sahel, J.-A., & Dalkara, D.
Affiliations
Centre National de la Recherche Scientifique (FR)
Corresponding author
Dalkara, Deniz
Venue
Progress in Retinal and Eye Research
Publisher
Elsevier BV
Published
2022-01
Volume
86
Pages
100975
ISSN
1350-9462 (ISSN-L)
OpenAlex
W3165170182
Cited by
190
References
183
Citations by year
2026: 24 · 2025: 47 · 2024: 31 · 2023: 54 · 2022: 26 · 2021: 7 · 2014: 1
FWCI
3.09
Type
journal article
Language
en
Open access
Locations
  • Progress in Retinal and Eye Research (publishedVersion, cc-by-nc-nd)
  • PubMed (publishedVersion)
  • HAL (submittedVersion, other-oa)
Funders
  • Île-de-France Regional Council
  • Foundation Fighting Blindness Inc
  • French National Research Agency
  • European Research Council
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Retinal Development and Disorders
  • Health Sciences › Medicine › Ophthalmology › Retinal Diseases and Treatments
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Virus-based gene therapy research
Keywords
retinal gene therapy, inherited retinal degeneration, retinitis pigmentosa, age-related macular degeneration, gene delivery vectors, genome editing, vision loss
MeSH terms
Humans, Macular Degeneration, Retina, Retinal Degeneration, Retinitis Pigmentosa, Genetic Therapy

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