ArticleInvestigative ophthalmology & visual science2013
Ocular phenotype of Fbn2-null mice.
Article in Investigative ophthalmology & visual science, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 27 papers.
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Who cites it
27 citing papers in PubMed, 32 citations in OpenAlex.
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- Altered Ocular Fibrillin Microfibril Composition in Mice With a Glaucoma-Causing Mutation of Adamts10.Investigative ophthalmology & visual science · 2021Article
- Nanophthalmos-Associated MYRF Gene Mutation Causes Ciliary Zonule Defects in Mice.Investigative ophthalmology & visual science · 2021Article
- A multiethnic genome-wide analysis of 44,039 individuals identifies 41 new loci associated with central corneal thickness.Communications biology · 2020Article
- An update on the genetics of ocular coloboma.Human genetics · 2019Review
- Adamts10 inactivation in mice leads to persistence of ocular microfibrils subsequent to reduced fibrillin-2 cleavage.Matrix biology : journal of the International Society for Matrix Biology · 2019Article
- Targeted deletion of fibrillin-1 in the mouse eye results in ectopia lentis and other ocular phenotypes associated with Marfan syndrome.Disease models & mechanisms · 2019Article
- ADAMTS10-mediated tissue disruption in Weill-Marchesani syndrome.Human molecular genetics · 2018Article
- Optimizing Genomic Methods for Mapping and Identification of Candidate Variants in ENU Mutagenesis Screens Using Inbred Mice.G3 (Bethesda, Md.) · 2018Article
- A child with Myhre syndrome presenting with corectopia and tetralogy of Fallot.American journal of medical genetics. Part A · 2018Article
- Nonproliferative and Proliferative Lesions of the Ratand Mouse Special Sense Organs(Ocular [eye and glands], Olfactory and Otic).Journal of toxicologic pathology · 2018Review
- Proteomic Analysis of the Bovine and Human Ciliary Zonule.Investigative ophthalmology & visual science · 2017Article
- A full lifespan model of vertebrate lens growth.Royal Society open science · 2017Article
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Authors and funding
4 authors at 1 institution in 1 country.
Funding
Abstract
purposeFibrillin-2 (Fbn2) is the dominant fibrillin isoform expressed during development of the mouse eye. To test its role in morphogenesis, we examined the ocular phenotype of Fbn2(-/-) mice.
methodsOcular morphology was assessed by confocal microscopy using antibodies against microfibril components.
resultsFbn2(-/-) mice had a high incidence of anterior segment dysgenesis. The iris was the most commonly affected tissue. Complete iridal coloboma was present in 37% of eyes. Dyscoria, corectopia and pseudopolycoria were also common (43% combined incidence). In wild-type (WT) mice, fibrillin-2-rich microfibrils are prominent in the pupillary membrane (PM) during development. In Fbn2-null mice, the absence of Fbn2 was partially compensated for by increased expression of fibrillin-1, although the resulting PM microfibrils were disorganized, compared with WTs. In colobomatous adult Fbn2(-/-) eyes, the PM failed to regress normally, especially beneath the notched region of the iris. Segments of the ciliary body were hypoplastic, and zonular fibers, although relatively plentiful, were unevenly distributed around the lens equator. In regions where the zonular fibers were particularly disturbed, the synchronous differentiation of the underlying lens fiber cells was affected.
conclusionsFbn2 has an indispensable role in ocular morphogenesis in mice. The high incidence of iris coloboma in Fbn2-null animals implies a previously unsuspected role in optic fissure closure. The observation that fiber cell differentiation was disturbed in Fbn2(-/-) mice raises the possibility that the attachment of zonular fibers to the lens surface may help specify the equatorial margin of the lens epithelium.
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