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Prelamin A causes aberrant myonuclear arrangement and results in muscle fiber weakness

Physiological and premature aging are frequently associated with an accumulation of prelamin A, a precursor of lamin A, in the nuclear envelope of various cell types. Here, we aimed to underpin the hitherto unknown mechanisms by which prelamin A alters myonuclear organization and muscle fiber functi...

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Autores principales: Levy, Yotam, Ross, Jacob A., Niglas, Marili, Snetkov, Vladimir A., Lynham, Steven, Liao, Chen-Yu, Puckelwartz, Megan J., Hsu, Yueh-Mei, McNally, Elizabeth M., Alsheimer, Manfred, Harridge, Stephen D.R., Young, Stephen G., Fong, Loren G., Español, Yaiza, Lopez-Otin, Carlos, Kennedy, Brian K., Lowe, Dawn A., Ochala, Julien
Formato: Online Artículo Texto
Lenguaje:English
Publicado: American Society for Clinical Investigation 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6237469/
https://www.ncbi.nlm.nih.gov/pubmed/30282816
http://dx.doi.org/10.1172/jci.insight.120920
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author Levy, Yotam
Ross, Jacob A.
Niglas, Marili
Snetkov, Vladimir A.
Lynham, Steven
Liao, Chen-Yu
Puckelwartz, Megan J.
Hsu, Yueh-Mei
McNally, Elizabeth M.
Alsheimer, Manfred
Harridge, Stephen D.R.
Young, Stephen G.
Fong, Loren G.
Español, Yaiza
Lopez-Otin, Carlos
Kennedy, Brian K.
Lowe, Dawn A.
Ochala, Julien
author_facet Levy, Yotam
Ross, Jacob A.
Niglas, Marili
Snetkov, Vladimir A.
Lynham, Steven
Liao, Chen-Yu
Puckelwartz, Megan J.
Hsu, Yueh-Mei
McNally, Elizabeth M.
Alsheimer, Manfred
Harridge, Stephen D.R.
Young, Stephen G.
Fong, Loren G.
Español, Yaiza
Lopez-Otin, Carlos
Kennedy, Brian K.
Lowe, Dawn A.
Ochala, Julien
author_sort Levy, Yotam
collection PubMed
description Physiological and premature aging are frequently associated with an accumulation of prelamin A, a precursor of lamin A, in the nuclear envelope of various cell types. Here, we aimed to underpin the hitherto unknown mechanisms by which prelamin A alters myonuclear organization and muscle fiber function. By experimentally studying membrane-permeabilized myofibers from various transgenic mouse lines, our results indicate that, in the presence of prelamin A, the abundance of nuclei and myosin content is markedly reduced within muscle fibers. This leads to a concept by which the remaining myonuclei are very distant from each other and are pushed to function beyond their maximum cytoplasmic capacity, ultimately inducing muscle fiber weakness.
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spelling pubmed-62374692018-11-21 Prelamin A causes aberrant myonuclear arrangement and results in muscle fiber weakness Levy, Yotam Ross, Jacob A. Niglas, Marili Snetkov, Vladimir A. Lynham, Steven Liao, Chen-Yu Puckelwartz, Megan J. Hsu, Yueh-Mei McNally, Elizabeth M. Alsheimer, Manfred Harridge, Stephen D.R. Young, Stephen G. Fong, Loren G. Español, Yaiza Lopez-Otin, Carlos Kennedy, Brian K. Lowe, Dawn A. Ochala, Julien JCI Insight Research Article Physiological and premature aging are frequently associated with an accumulation of prelamin A, a precursor of lamin A, in the nuclear envelope of various cell types. Here, we aimed to underpin the hitherto unknown mechanisms by which prelamin A alters myonuclear organization and muscle fiber function. By experimentally studying membrane-permeabilized myofibers from various transgenic mouse lines, our results indicate that, in the presence of prelamin A, the abundance of nuclei and myosin content is markedly reduced within muscle fibers. This leads to a concept by which the remaining myonuclei are very distant from each other and are pushed to function beyond their maximum cytoplasmic capacity, ultimately inducing muscle fiber weakness. American Society for Clinical Investigation 2018-10-04 /pmc/articles/PMC6237469/ /pubmed/30282816 http://dx.doi.org/10.1172/jci.insight.120920 Text en Copyright © 2018 Levy et al. http://creativecommons.org/licenses/by/4.0/ This work is licensed under the Creative Commons Attribution 4.0 International License.
spellingShingle Research Article
Levy, Yotam
Ross, Jacob A.
Niglas, Marili
Snetkov, Vladimir A.
Lynham, Steven
Liao, Chen-Yu
Puckelwartz, Megan J.
Hsu, Yueh-Mei
McNally, Elizabeth M.
Alsheimer, Manfred
Harridge, Stephen D.R.
Young, Stephen G.
Fong, Loren G.
Español, Yaiza
Lopez-Otin, Carlos
Kennedy, Brian K.
Lowe, Dawn A.
Ochala, Julien
Prelamin A causes aberrant myonuclear arrangement and results in muscle fiber weakness
title Prelamin A causes aberrant myonuclear arrangement and results in muscle fiber weakness
title_full Prelamin A causes aberrant myonuclear arrangement and results in muscle fiber weakness
title_fullStr Prelamin A causes aberrant myonuclear arrangement and results in muscle fiber weakness
title_full_unstemmed Prelamin A causes aberrant myonuclear arrangement and results in muscle fiber weakness
title_short Prelamin A causes aberrant myonuclear arrangement and results in muscle fiber weakness
title_sort prelamin a causes aberrant myonuclear arrangement and results in muscle fiber weakness
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6237469/
https://www.ncbi.nlm.nih.gov/pubmed/30282816
http://dx.doi.org/10.1172/jci.insight.120920
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