Knockout of zebrafish desmin genes does not cause skeletal muscle degeneration but alters calcium flux

Abstract Desmin is a muscle-specific intermediate filament protein that has fundamental role in muscle structure and force transmission. Whereas human desmin protein is encoded by a single gene, two desmin paralogs (desma and desmb) exist in zebrafish. Desma and desmb show differential spatiotempora...

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Autores principales:	Gülsüm Kayman Kürekçi, Ecem Kural Mangit, Cansu Koyunlar, Seyda Unsal, Berk Saglam, Bora Ergin, Merve Gizer, Ismail Uyanik, Niloufar Boustanabadimaralan Düz, Petek Korkusuz, Beril Talim, Nuhan Purali, Simon M. Hughes, Pervin R. Dincer
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2021
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/55f94f846ead4d2a8d2457a327731aba
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spelling	oai:doaj.org-article:55f94f846ead4d2a8d2457a327731aba2021-12-02T14:20:43ZKnockout of zebrafish desmin genes does not cause skeletal muscle degeneration but alters calcium flux10.1038/s41598-021-86974-w2045-2322https://doaj.org/article/55f94f846ead4d2a8d2457a327731aba2021-04-01T00:00:00Zhttps://doi.org/10.1038/s41598-021-86974-whttps://doaj.org/toc/2045-2322Abstract Desmin is a muscle-specific intermediate filament protein that has fundamental role in muscle structure and force transmission. Whereas human desmin protein is encoded by a single gene, two desmin paralogs (desma and desmb) exist in zebrafish. Desma and desmb show differential spatiotemporal expression during zebrafish embryonic and larval development, being similarly expressed in skeletal muscle until hatching, after which expression of desmb shifts to gut smooth muscle. We generated knockout (KO) mutant lines carrying loss-of-function mutations for each gene by using CRISPR/Cas9. Mutants are viable and fertile, and lack obvious skeletal muscle, heart or intestinal defects. In contrast to morphants, knockout of each gene did not cause any overt muscular phenotype, but did alter calcium flux in myofibres. These results point to a possible compensation mechanism in these mutant lines generated by targeting nonsense mutations to the first coding exon.Gülsüm Kayman KürekçiEcem Kural MangitCansu KoyunlarSeyda UnsalBerk SaglamBora ErginMerve GizerIsmail UyanikNiloufar Boustanabadimaralan DüzPetek KorkusuzBeril TalimNuhan PuraliSimon M. HughesPervin R. DincerNature PortfolioarticleMedicineRScienceQENScientific Reports, Vol 11, Iss 1, Pp 1-15 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Gülsüm Kayman Kürekçi Ecem Kural Mangit Cansu Koyunlar Seyda Unsal Berk Saglam Bora Ergin Merve Gizer Ismail Uyanik Niloufar Boustanabadimaralan Düz Petek Korkusuz Beril Talim Nuhan Purali Simon M. Hughes Pervin R. Dincer Knockout of zebrafish desmin genes does not cause skeletal muscle degeneration but alters calcium flux
description	Abstract Desmin is a muscle-specific intermediate filament protein that has fundamental role in muscle structure and force transmission. Whereas human desmin protein is encoded by a single gene, two desmin paralogs (desma and desmb) exist in zebrafish. Desma and desmb show differential spatiotemporal expression during zebrafish embryonic and larval development, being similarly expressed in skeletal muscle until hatching, after which expression of desmb shifts to gut smooth muscle. We generated knockout (KO) mutant lines carrying loss-of-function mutations for each gene by using CRISPR/Cas9. Mutants are viable and fertile, and lack obvious skeletal muscle, heart or intestinal defects. In contrast to morphants, knockout of each gene did not cause any overt muscular phenotype, but did alter calcium flux in myofibres. These results point to a possible compensation mechanism in these mutant lines generated by targeting nonsense mutations to the first coding exon.
format	article
author	Gülsüm Kayman Kürekçi Ecem Kural Mangit Cansu Koyunlar Seyda Unsal Berk Saglam Bora Ergin Merve Gizer Ismail Uyanik Niloufar Boustanabadimaralan Düz Petek Korkusuz Beril Talim Nuhan Purali Simon M. Hughes Pervin R. Dincer
author_facet	Gülsüm Kayman Kürekçi Ecem Kural Mangit Cansu Koyunlar Seyda Unsal Berk Saglam Bora Ergin Merve Gizer Ismail Uyanik Niloufar Boustanabadimaralan Düz Petek Korkusuz Beril Talim Nuhan Purali Simon M. Hughes Pervin R. Dincer
author_sort	Gülsüm Kayman Kürekçi
title	Knockout of zebrafish desmin genes does not cause skeletal muscle degeneration but alters calcium flux
title_short	Knockout of zebrafish desmin genes does not cause skeletal muscle degeneration but alters calcium flux
title_full	Knockout of zebrafish desmin genes does not cause skeletal muscle degeneration but alters calcium flux
title_fullStr	Knockout of zebrafish desmin genes does not cause skeletal muscle degeneration but alters calcium flux
title_full_unstemmed	Knockout of zebrafish desmin genes does not cause skeletal muscle degeneration but alters calcium flux
title_sort	knockout of zebrafish desmin genes does not cause skeletal muscle degeneration but alters calcium flux
publisher	Nature Portfolio
publishDate	2021
url	https://doaj.org/article/55f94f846ead4d2a8d2457a327731aba
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Knockout of zebrafish desmin genes does not cause skeletal muscle degeneration but alters calcium flux

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