ArticleDevelopmental biology2007
Defects in eye development in transgenic mice overexpressing the heparan sulfate proteoglycan agrin.
Article in Developmental biology, 2007. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 22 papers.
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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.
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Who cites it
22 citing papers in PubMed, 41 citations in OpenAlex.
- Genetic variants in genes regulating lens epithelial cell homeostasis in dead bag syndrome: part 2.Journal of cataract and refractive surgery · 2026Article
- Spatiotemporal Localisation of Heparan Sulphate Proteoglycans throughout Mouse Lens Morphogenesis.Cells · 2023Article
- Single-cell RNA sequencing of the retina in a model of retinitis pigmentosa reveals early responses to degeneration in rods and cones.BMC biology · 2022Article
- Ehlers-Danlos syndromes and their manifestations in the visual system.Frontiers in medicine · 2022Review
- An Atlas of Heparan Sulfate Proteoglycans in the Postnatal Rat Lens.Investigative ophthalmology & visual science · 2021Article
- Distribution and Function of Glycosaminoglycans and Proteoglycans in the Development, Homeostasis and Pathology of the Ocular Surface.Frontiers in cell and developmental biology · 2020Review
- Transformation of the Transcriptomic Profile of Mouse Periocular Mesenchyme During Formation of the Embryonic Cornea.Investigative ophthalmology & visual science · 2019Article
- Micromorphology analysis of the anterior human lens capsule.Molecular vision · 2018Article
- The role of heparan sulphate in development: the ectodermal story.International journal of experimental pathology · 2016Review
- Strain-Dependent Anterior Segment Dysgenesis and Progression to Glaucoma in Col4a1 Mutant Mice.Investigative ophthalmology & visual science · 2015Article
- Lens capsule structure assessed with atomic force microscopy.Molecular vision · 2015Article
- Stem cell therapy of cataract.BioImpacts : BI · 2015Article
- Extracellular Matrix is an Important Component of Limbal Stem Cell Niche.Journal of functional biomaterials · 2012Article
- Article
- A valid mouse model of AGRIN-associated congenital myasthenic syndrome.Human molecular genetics · 2011Article
- Changes in brain β-amyloid deposition and aquaporin 4 levels in response to altered agrin expression in mice.Journal of neuropathology and experimental neurology · 2011Article
- Agrin function associated with ocular development is a target of ethanol exposure in embryonic zebrafish.Birth defects research. Part A, Clinical and molecular teratology · 2011Article
- A novel null allele of mouse DSCAM survives to adulthood on an inbred C3H background with reduced phenotypic variability.Genesis (New York, N.Y. : 2000) · 2010Article
- DSCAM and DSCAML1 function in self-avoidance in multiple cell types in the developing mouse retina.Neuron · 2009Article
- Incomplete penetrance and phenotypic variability characterize Gdf6-attributable oculo-skeletal phenotypes.Human molecular genetics · 2009Article
Corrections and comments
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Authors and funding
3 authors at 1 institution in 1 country.
Funding
Abstract
The importance of heparan sulfate proteoglycans (HSPGs) in neurodevelopment is becoming increasingly clear. However, studies on HSPGs are hampered by pleiotropic effects when synthesis or modification of heparan sulfate itself is targeted, and by redundancy when the core proteins are altered. Gain-of-function experiments can sometimes circumvent these issues. Here we establish that transgenic mice overexpressing the HSPG agrin have severe ocular dysgenesis. The defects occur through a gain-of-function mechanism and penetrance is dependent on agrin dosage. The agrin-induced developmental defects are highly variable, and include anophthalmia, persistence of vitreous vessels, and fusion of anterior chamber structures. A frequently observed defect is an optic stalk coloboma leading to the misdifferentiation of the optic stalk as retina, which becomes continuous with the forebrain. The defects in optic-stalk differentiation correlate with reduced sonic hedgehog immunoreactivity and overexpansion of the PAX6 domain from the retina into the optic stalk. The ocular phenotypes associated with agrin overexpression are dependent on genetic background, occurring with high penetrance in inbred C57BL/6J mice. Distinct loci sensitizing C57BL/6J mice to agrin-induced dysgenesis were identified. These results indicate that agrin overexpression will provide a tool to explore the molecular interactions of the extracellular matrix and cell surface in eye development, and provide a means for identifying modifier loci that sensitize mice to developmental eye defects.
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Registered trials
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.