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PMID: 9050868 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Type III collagen is crucial for collagen I fibrillogenesis and for normal cardiovascular development.

Liu X, Wu H, Byrne M, Krane S, Jaenisch R

Abstract

Type III collagen is a fibrillar forming collagen comprising three alpha1(III) chains and is expressed in early embryos and throughout embryogenesis. In the adult, type III collagen is a major component of the extracellular matrix in a variety of internal organs and skin. Mutations in the COL3A1 gene have been implicated as a cause of type IV Ehlers-Danlos syndrome, a disease leading to aortic rupture in early adult life. To directly study the role of Col3a1 in development and disease, we have inactivated the Col3a1 gene in embryonic stem cells by homologous recombination. The mutated allele was transmitted through the mouse germ line and homozygous mutant animals were derived from heterozygous intercrosses. About 10% of the homozygous mutant animals survived to adulthood but have a much shorter life span compared with wild-type mice. The major cause of death of mutant mice was rupture of the major blood vessels, similar to patients with type IV Ehlers-Danlos syndrome. Ultrastructural analysis of tissues from mutant mice revealed that type III collagen is essential for normal collagen I fibrillogenesis in the cardiovascular system and other organs.

MeSH Terms
Animals Aorta/pathology,ultrastructure Blotting, Southern Cardiovascular System/growth & development,metabolism Cells, Cultured Collagen/genetics,metabolism Disease Models, Animal Ehlers-Danlos Syndrome/genetics,pathology,physiopathology Gene Targeting Histocytochemistry Mice Mice, Transgenic Microscopy, Electron Myocardium/pathology,ultrastructure Restriction Mapping Sequence Deletion/genetics Skin/ultrastructure Stem Cells
Chemicals
Collagen
Authors & Affiliations
5 authors, click to expand affiliations / ORCID
Liu X
The Whitehead Institute for Biomedical Research, Massachusetts Institute of Technology, Cambridge 02142, USA.
Wu H
Byrne M
Krane S
Jaenisch R
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Article Info
Journal
Proceedings of the National Academy of Sciences of the United States of America
Abbr.
Proc Natl Acad Sci U S A
ISSN
0027-8424
Published
1997-03-04
Pages
1852-6
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC20006
Subset
IM
Grants
NIAMS NIH HHS · AR-3564 · United States
NCI NIH HHS · R35-CA44339 · United States
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