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Dysferlin-deficient immortalized human myoblasts and myotubes as a useful tool to study dysferlinopathy
Dysferlin gene mutations causing LGMD2B are associated with defects in muscle membrane repair. Four stable cell lines have been established from primary human dysferlin-deficient myoblasts harbouring different mutations in the dysferlin gene. We have compared immortalized human myoblasts and myotube...
Autores principales: | , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Public Library of Science
2012
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3274833/ https://www.ncbi.nlm.nih.gov/pubmed/22367358 http://dx.doi.org/10.1371/currents.RRN1298 |
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author | Philippi, Susanne Bigot, Anne Marg, Andreas Mouly, Vincent Spuler, Simone Zacharias, Ute |
author_facet | Philippi, Susanne Bigot, Anne Marg, Andreas Mouly, Vincent Spuler, Simone Zacharias, Ute |
author_sort | Philippi, Susanne |
collection | PubMed |
description | Dysferlin gene mutations causing LGMD2B are associated with defects in muscle membrane repair. Four stable cell lines have been established from primary human dysferlin-deficient myoblasts harbouring different mutations in the dysferlin gene. We have compared immortalized human myoblasts and myotubes carrying disease-causing mutations in dysferlin to their wild-type counterparts. Fusion of myoblasts into myotubes and expression of muscle-specific differentiation markers were investigated with special emphasis on dysferlin protein expression, subcellular localization and function in membrane repair. We found that the immortalized myoblasts and myotubes were virtually indistinguishable from their parental cell line for all of the criteria we investigated. They therefore will provide a very useful tool to further investigate dysferlin function and pathophysiology as well as to test therapeutic strategies at the cellular level. |
format | Online Article Text |
id | pubmed-3274833 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2012 |
publisher | Public Library of Science |
record_format | MEDLINE/PubMed |
spelling | pubmed-32748332012-02-23 Dysferlin-deficient immortalized human myoblasts and myotubes as a useful tool to study dysferlinopathy Philippi, Susanne Bigot, Anne Marg, Andreas Mouly, Vincent Spuler, Simone Zacharias, Ute PLoS Curr Muscular Dystrophy Dysferlin gene mutations causing LGMD2B are associated with defects in muscle membrane repair. Four stable cell lines have been established from primary human dysferlin-deficient myoblasts harbouring different mutations in the dysferlin gene. We have compared immortalized human myoblasts and myotubes carrying disease-causing mutations in dysferlin to their wild-type counterparts. Fusion of myoblasts into myotubes and expression of muscle-specific differentiation markers were investigated with special emphasis on dysferlin protein expression, subcellular localization and function in membrane repair. We found that the immortalized myoblasts and myotubes were virtually indistinguishable from their parental cell line for all of the criteria we investigated. They therefore will provide a very useful tool to further investigate dysferlin function and pathophysiology as well as to test therapeutic strategies at the cellular level. Public Library of Science 2012-02-28 /pmc/articles/PMC3274833/ /pubmed/22367358 http://dx.doi.org/10.1371/currents.RRN1298 Text en http://creativecommons.org/licenses/by/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are properly credited. |
spellingShingle | Muscular Dystrophy Philippi, Susanne Bigot, Anne Marg, Andreas Mouly, Vincent Spuler, Simone Zacharias, Ute Dysferlin-deficient immortalized human myoblasts and myotubes as a useful tool to study dysferlinopathy |
title | Dysferlin-deficient immortalized human myoblasts and myotubes as a useful tool to study dysferlinopathy |
title_full | Dysferlin-deficient immortalized human myoblasts and myotubes as a useful tool to study dysferlinopathy |
title_fullStr | Dysferlin-deficient immortalized human myoblasts and myotubes as a useful tool to study dysferlinopathy |
title_full_unstemmed | Dysferlin-deficient immortalized human myoblasts and myotubes as a useful tool to study dysferlinopathy |
title_short | Dysferlin-deficient immortalized human myoblasts and myotubes as a useful tool to study dysferlinopathy |
title_sort | dysferlin-deficient immortalized human myoblasts and myotubes as a useful tool to study dysferlinopathy |
topic | Muscular Dystrophy |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3274833/ https://www.ncbi.nlm.nih.gov/pubmed/22367358 http://dx.doi.org/10.1371/currents.RRN1298 |
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