ArticleBMC neuroscience2005
Synaptogenesis and outer segment formation are perturbed in the neural retina of Crx mutant mice.
Article in BMC neuroscience, 2005. 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, 64 citations in OpenAlex.
- Lineage tracing reveals photoreceptor precursor cell subpopulations that contribute to murine retinogenesis.Frontiers in cell and developmental biology · 2026Article
- USH2A-Mutated Human Retinal Organoids Model Rod-Cone Dystrophy.Investigative ophthalmology & visual science · 2025Article
- SNAP-25, but not SNAP-23, is essential for photoreceptor development, survival, and function in mice.Communications biology · 2024Article
- Progenitor division and cell autonomous neurosecretion are required for rod photoreceptor sublaminar positioning.Proceedings of the National Academy of Sciences of the United States of America · 2023Article
- Cat LCA-CRX Model, Homozygous for an Antimorphic Mutation Has a Unique Phenotype.Translational vision science & technology · 2023Article
- Activity-dependent regulation of mitochondrial motility in developing cortical dendrites.eLife · 2021Article
- Inner retinal preservation in the photoinducible I307N rhodopsin mutant mouse, a model of autosomal dominant retinitis pigmentosa.The Journal of comparative neurology · 2020Article
- Review
- Inherited Eye Diseases with Retinal Manifestations through the Eyes of Homeobox Genes.International journal of molecular sciences · 2020Review
- Crx Is Posttranscriptionally Regulated by Light Stimulation in Postnatal Rat Retina.Frontiers in cell and developmental biology · 2020Article
- Microglia Mediate Synaptic Material Clearance at the Early Stage of Rats With Retinitis Pigmentosa.Frontiers in immunology · 2019Article
- Rpgrip1 is required for rod outer segment development and ciliary protein trafficking in zebrafish.Scientific reports · 2017Article
- Insights from Genetic Model Systems of Retinal Degeneration: Role of Epsins in Retinal Angiogenesis and VEGFR2 Signaling.Journal of nature and science · 2017Article
- The Musashi 1 Controls the Splicing of Photoreceptor-Specific Exons in the Vertebrate Retina.PLoS genetics · 2016Article
- CrxRdy Cat: A Large Animal Model for CRX-Associated Leber Congenital Amaurosis.Investigative ophthalmology & visual science · 2016Article
- Orthodenticle Is Required for the Expression of Principal Recognition Molecules That Control Axon Targeting in the Drosophila Retina.PLoS genetics · 2015Article
- Synaptic remodeling of neuronal circuits in early retinal degeneration.Frontiers in cellular neuroscience · 2015Review
- Mechanisms of blindness: animal models provide insight into distinct CRX-associated retinopathies.Developmental dynamics : an official publication of the American Association of Anatomists · 2014Review
- Mechanistically distinct mouse models for CRX-associated retinopathy.PLoS genetics · 2014Article
- Conserved roles of ems/Emx and otd/Otx genes in olfactory and visual system development in Drosophila and mouse.Open biology · 2013Review
Corrections and comments
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Authors and funding
4 authors at 2 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundIn Leber's congenital amaurosis (LCA), affected individuals are blind, or nearly so, from birth. This early onset suggests abnormal development of the neural retina. Mutations in genes that affect the development and/or function of photoreceptor cells have been found to be responsible in some families. These examples include mutations in the photoreceptor transcription factor, Crx.
resultsA Crx mutant strain of mice was created to serve as a model for LCA and to provide more insight into Crx's function. In this study, an ultrastructural analysis of the developing retina in Crx mutant mice was performed. Outer segment morphogenesis was found to be blocked at the elongation stage, leading to a failure in production of the phototransduction apparatus. Further, Crx-/- photoreceptors demonstrated severely abnormal synaptic endings in the outer plexiform layer.
conclusionsThis is the first report of a synaptogenesis defect in an animal model for LCA. These data confirm the essential role this gene plays in multiple aspects of photoreceptor development and extend our understanding of the basic pathology of LCA.
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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.