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...
Autores principales: | , , , , |
---|---|
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 |
_version_ | 1782152584466792448 |
---|---|
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. |
format | Text |
id | pubmed-2315682 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2008 |
publisher | The Rockefeller University Press |
record_format | MEDLINE/PubMed |
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 |
work_keys_str_mv | AT zoellerjasonj acentralfunctionforperlecaninskeletalmuscleandcardiovasculardevelopment AT mcquillanangela acentralfunctionforperlecaninskeletalmuscleandcardiovasculardevelopment AT whitelockjohn acentralfunctionforperlecaninskeletalmuscleandcardiovasculardevelopment AT hoshiuying acentralfunctionforperlecaninskeletalmuscleandcardiovasculardevelopment AT iozzorenatov acentralfunctionforperlecaninskeletalmuscleandcardiovasculardevelopment AT zoellerjasonj centralfunctionforperlecaninskeletalmuscleandcardiovasculardevelopment AT mcquillanangela centralfunctionforperlecaninskeletalmuscleandcardiovasculardevelopment AT whitelockjohn centralfunctionforperlecaninskeletalmuscleandcardiovasculardevelopment AT hoshiuying centralfunctionforperlecaninskeletalmuscleandcardiovasculardevelopment AT iozzorenatov centralfunctionforperlecaninskeletalmuscleandcardiovasculardevelopment |