Linked Life Data

19782070

Source:http://linkedlifedata.com/resource/pubmed/id/19782070

Statements in which the resource exists as a subject.
PredicateObject
rdf:type
lifeskim:mentions
pubmed:issue
1
pubmed:dateCreated
2009-12-4
pubmed:abstractText
The heparan sulfate (HS) is a component of proteoglycans in the extracellular matrix and on cell surfaces, modulating developmental processes. The aim of this study is to investigate whether the defect of HS in the periocular mesenchyme impairs ocular morphogenesis. First, using Protein 0-Cre transgenic mice, we ablated Ext1, which encodes an indispensable enzyme for HS synthesis, in the developing periocular mesenchyme. The expression of Ext1 messenger RNA (mRNA) and HS were observed by RT-PCR and immunohistochemistry, respectively. The phenotypes in the mutant were evaluated by light microscopy and immunohistochemistry for cellular makers. Second, the distribution of the mutant periocular mesenchymal cells was tracked using a Rosa26 Cre-reporter gene. No mutant embryos (Protein 0-Cre;Ext1(flox/flox)) were identified after embryonic day 14.5 (E14.5). RT-PCR showed that an intense band amplified from Ext1 was observed in cDNAs from the control periocular mesenchymal cells at E13.5; however, the band for Ext1 was hardly detectable in cDNA from the mutant embryo, indicating that the mRNA was missing in the mutant periocular mesenchyme at E13.5. The HS expression was disrupted in the periocular mesenchyme of the mutant ocular tissues. The HS deficiency resulted in microphthalmia with reduced axial lengths, lens diameters, and vitreous sizes compared with the littermate eyes. The mutant embryos showed agenesis of the anterior chamber, where cells expressing Cre recombinase were distributed. Moreover, the mutants showed phenotypic alterations in the neural ectoderm including dysgenesis of the presumptive ciliary body and agenesis of the optic nerve head. These findings demonstrate that HS in the periocular mesenchyme plays a critical role in normal ocular morphogenesis, indicating reciprocal interactions between the periocular mesenchyme and the neural ectoderm.
pubmed:language
eng
pubmed:journal
pubmed:citationSubset
IM
pubmed:chemical
pubmed:status
MEDLINE
pubmed:month
Jan
pubmed:issn
1096-0007
pubmed:author
pubmed:issnType
Electronic
pubmed:volume
90
pubmed:owner
NLM
pubmed:authorsComplete
Y
pubmed:pagination
81-8
pubmed:meshHeading
pubmed-meshheading:19782070-Animals, pubmed-meshheading:19782070-Carbohydrate Epimerases, pubmed-meshheading:19782070-Ciliary Body, pubmed-meshheading:19782070-Eye Abnormalities, pubmed-meshheading:19782070-Female, pubmed-meshheading:19782070-Genotype, pubmed-meshheading:19782070-Heparitin Sulfate, pubmed-meshheading:19782070-Immunoenzyme Techniques, pubmed-meshheading:19782070-Male, pubmed-meshheading:19782070-Mesoderm, pubmed-meshheading:19782070-Mice, pubmed-meshheading:19782070-Mice, Transgenic, pubmed-meshheading:19782070-Microphthalmos, pubmed-meshheading:19782070-Morphogenesis, pubmed-meshheading:19782070-N-Acetylglucosaminyltransferases, pubmed-meshheading:19782070-RNA, Messenger, pubmed-meshheading:19782070-Reverse Transcriptase Polymerase Chain Reaction, pubmed-meshheading:19782070-Smad Proteins, pubmed-meshheading:19782070-Sulfotransferases
pubmed:year
2010
pubmed:articleTitle
Heparan sulfate deficiency in periocular mesenchyme causes microphthalmia and ciliary body dysgenesis.
pubmed:affiliation
Department of Ophthalmology and Visual Science, Kumamoto University, Graduate School of Medical Sciences, 1-1-1, Honjo, 860-8556 Kumamoto City, Japan.
pubmed:publicationType
Journal Article, Research Support, Non-U.S. Gov't