A central function for perlecan in skeletal muscle and cardiovascular development

Perlecan's developmental functions are difficult to dissect in placental animals because perlecan disruption is embryonic lethal. In contrast to mammals, cardiovascular function is not essential for early zebrafish development because the embryos obtain adequate oxygen by diffusion. In this stu...

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Autores principales: Zoeller, Jason J., McQuillan, Angela, Whitelock, John, Ho, Shiu-Ying, Iozzo, Renato V.
Formato: Texto
Lenguaje:English
Publicado: The Rockefeller University Press 2008
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2315682/
https://www.ncbi.nlm.nih.gov/pubmed/18426981
http://dx.doi.org/10.1083/jcb.200708022
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author Zoeller, Jason J.
McQuillan, Angela
Whitelock, John
Ho, Shiu-Ying
Iozzo, Renato V.
author_facet Zoeller, Jason J.
McQuillan, Angela
Whitelock, John
Ho, Shiu-Ying
Iozzo, Renato V.
author_sort Zoeller, Jason J.
collection PubMed
description Perlecan's developmental functions are difficult to dissect in placental animals because perlecan disruption is embryonic lethal. In contrast to mammals, cardiovascular function is not essential for early zebrafish development because the embryos obtain adequate oxygen by diffusion. In this study, we use targeted protein depletion coupled with protein-based rescue experiments to investigate the involvement of perlecan and its C-terminal domain V/endorepellin in zebrafish development. The perlecan morphants show a severe myopathy characterized by abnormal actin filament orientation and disorganized sarcomeres, suggesting an involvement of perlecan in myopathies. In the perlecan morphants, primary intersegmental vessel sprouts, which develop through angiogenesis, fail to extend and show reduced protrusive activity. Live videomicroscopy confirms the abnormal swimming pattern caused by the myopathy and anomalous head and trunk vessel circulation. The phenotype is partially rescued by microinjection of human perlecan or endorepellin. These findings indicate that perlecan is essential for the integrity of somitic muscle and developmental angiogenesis and that endorepellin mediates most of these biological activities.
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spelling pubmed-23156822008-10-21 A central function for perlecan in skeletal muscle and cardiovascular development Zoeller, Jason J. McQuillan, Angela Whitelock, John Ho, Shiu-Ying Iozzo, Renato V. J Cell Biol Research Articles Perlecan's developmental functions are difficult to dissect in placental animals because perlecan disruption is embryonic lethal. In contrast to mammals, cardiovascular function is not essential for early zebrafish development because the embryos obtain adequate oxygen by diffusion. In this study, we use targeted protein depletion coupled with protein-based rescue experiments to investigate the involvement of perlecan and its C-terminal domain V/endorepellin in zebrafish development. The perlecan morphants show a severe myopathy characterized by abnormal actin filament orientation and disorganized sarcomeres, suggesting an involvement of perlecan in myopathies. In the perlecan morphants, primary intersegmental vessel sprouts, which develop through angiogenesis, fail to extend and show reduced protrusive activity. Live videomicroscopy confirms the abnormal swimming pattern caused by the myopathy and anomalous head and trunk vessel circulation. The phenotype is partially rescued by microinjection of human perlecan or endorepellin. These findings indicate that perlecan is essential for the integrity of somitic muscle and developmental angiogenesis and that endorepellin mediates most of these biological activities. The Rockefeller University Press 2008-04-21 /pmc/articles/PMC2315682/ /pubmed/18426981 http://dx.doi.org/10.1083/jcb.200708022 Text en Copyright © 2008, The Rockefeller University Press This article is distributed under the terms of an Attribution–Noncommercial–Share Alike–No Mirror Sites license for the first six months after the publication date (see http://www.rupress.org/terms). After six months it is available under a Creative Commons License (Attribution–Noncommercial–Share Alike 4.0 Unported license, as described at http://creativecommons.org/licenses/by-nc-sa/4.0/).
spellingShingle Research Articles
Zoeller, Jason J.
McQuillan, Angela
Whitelock, John
Ho, Shiu-Ying
Iozzo, Renato V.
A central function for perlecan in skeletal muscle and cardiovascular development
title A central function for perlecan in skeletal muscle and cardiovascular development
title_full A central function for perlecan in skeletal muscle and cardiovascular development
title_fullStr A central function for perlecan in skeletal muscle and cardiovascular development
title_full_unstemmed A central function for perlecan in skeletal muscle and cardiovascular development
title_short A central function for perlecan in skeletal muscle and cardiovascular development
title_sort central function for perlecan in skeletal muscle and cardiovascular development
topic Research Articles
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2315682/
https://www.ncbi.nlm.nih.gov/pubmed/18426981
http://dx.doi.org/10.1083/jcb.200708022
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