ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience2015
A new CRB1 rat mutation links Müller glial cells to retinal telangiectasia.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 34 papers.
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Who cites it
34 citing papers in PubMed, 63 citations in OpenAlex.
- Expanding the Clinical Spectrum ofInternational journal of molecular sciences · 2025Article
- Genetic Background of Macular Telangiectasia Type 2.International journal of molecular sciences · 2025Review
- Multilayer Retinal Correspondence of the Structural and Vascular Anomalies in Eyes With Early Macular Telangiectasia Type 2.Investigative ophthalmology & visual science · 2024Article
- Diverse functions and pathogenetic role of Crumbs in retinopathy.Cell communication and signaling : CCS · 2024Review
- Relation of interdigitation zone changes and right-angle vessels in Macular Telangiectasia Type-2 (MacTel).Eye (London, England) · 2023Article
- Hyperpigmented epiretinal membrane in macular telangiectasia type 2: imaging characteristics and correlation with transretinal pigment migration.Eye (London, England) · 2023Article
- Characteristics of retinal pigment clumps in Type 2 macular telangiectasia (MacTel).Eye (London, England) · 2023Article
- HBEGF-TNF induce a complex outer retinal pathology with photoreceptor cell extrusion in human organoids.Nature communications · 2022Article
- Identification of Arhgef12 and Prkci as genetic modifiers of retinal dysplasia in the Crb1rd8 mouse model.PLoS genetics · 2022Article
- Dilated retinal large vessels and capillaries associated with diabetic macular edema and photoreceptor loss respectively.Graefe's archive for clinical and experimental ophthalmology = Albrecht von Graefes Archiv fur klinische und experimentelle Ophthalmologie · 2021Article
- Defining inclusion criteria and endpoints for clinical trials: a prospective cross-sectional study in CRB1-associated retinal dystrophies.Acta ophthalmologica · 2021Article
- Defining Phenotype, Tropism, and Retinal Gene Therapy Using Adeno-Associated Viral Vectors (AAVs) in New-Born Brown Norway Rats with a Spontaneous Mutation inInternational journal of molecular sciences · 2021Article
- AAV-Molecular therapy. Methods & clinical development · 2021Article
- Simplex Crumbs Homologue 1 Maculopathy Masquerading as Juvenile X-Linked Retinoschisis in Male Patients.Journal of vitreoretinal diseases · 2020Article
- Zebrafish Crb1, Localizing Uniquely to the Cell Membranes around Cone Photoreceptor Axonemes, Alleviates Light Damage to Photoreceptors and Modulates Cones' Light Responsiveness.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2020Article
- Rat models of human diseases and related phenotypes: a systematic inventory of the causative genes.Journal of biomedical science · 2020Review
- Transscleral Optical Phase Imaging of the Human Retina.Nature photonics · 2020Article
- AAV Targeting of Glial Cell Types in the Central and Peripheral Nervous System and Relevance to Human Gene Therapy.Frontiers in molecular neuroscience · 2020Review
- Research Models and Gene Augmentation Therapy forFrontiers in neuroscience · 2020Review
- Functional Genomics of the Retina to Elucidate its Construction and Deconstruction.International journal of molecular sciences · 2019Review
Corrections and comments
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Authors and funding
20 authors at 6 institutions in 5 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
We have identified and characterized a spontaneous Brown Norway from Janvier rat strain (BN-J) presenting a progressive retinal degeneration associated with early retinal telangiectasia, neuronal alterations, and loss of retinal Müller glial cells resembling human macular telangiectasia type 2 (MacTel 2), which is a retinal disease of unknown cause. Genetic analyses showed that the BN-J phenotype results from an autosomal recessive indel novel mutation in the Crb1 gene, causing dislocalization of the protein from the retinal Müller glia (RMG)/photoreceptor cell junction. The transcriptomic analyses of primary RMG cultures allowed identification of the dysregulated pathways in BN-J rats compared with wild-type BN rats. Among those pathways, TGF-β and Kit Receptor Signaling, MAPK Cascade, Growth Factors and Inflammatory Pathways, G-Protein Signaling Pathways, Regulation of Actin Cytoskeleton, and Cardiovascular Signaling were found. Potential molecular targets linking RMG/photoreceptor interaction with the development of retinal telangiectasia are identified. This model can help us to better understand the physiopathologic mechanisms of MacTel 2 and other retinal diseases associated with telangiectasia.
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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.