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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...

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Autores principales: Philippi, Susanne, Bigot, Anne, Marg, Andreas, Mouly, Vincent, Spuler, Simone, Zacharias, Ute
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Public Library of Science 2012
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.
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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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