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Analysis of RNA-Seq datasets reveals enrichment of tissue-specific splice variants for nuclear envelope proteins

Laminopathies yield tissue-specific pathologies, yet arise from mutation of ubiquitously-expressed genes. A little investigated hypothesis to explain this is that the mutated proteins or their partners have tissue-specific splice variants. To test this, we analyzed RNA-Seq datasets, finding novel is...

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Autores principales: Capitanchik, Charlotte, Dixon, Charles R., Swanson, Selene K., Florens, Laurence, Kerr, Alastair R. W., Schirmer, Eric C.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Taylor & Francis 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7000147/
https://www.ncbi.nlm.nih.gov/pubmed/29912636
http://dx.doi.org/10.1080/19491034.2018.1469351
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author Capitanchik, Charlotte
Dixon, Charles R.
Swanson, Selene K.
Florens, Laurence
Kerr, Alastair R. W.
Schirmer, Eric C.
author_facet Capitanchik, Charlotte
Dixon, Charles R.
Swanson, Selene K.
Florens, Laurence
Kerr, Alastair R. W.
Schirmer, Eric C.
author_sort Capitanchik, Charlotte
collection PubMed
description Laminopathies yield tissue-specific pathologies, yet arise from mutation of ubiquitously-expressed genes. A little investigated hypothesis to explain this is that the mutated proteins or their partners have tissue-specific splice variants. To test this, we analyzed RNA-Seq datasets, finding novel isoforms or isoform tissue-specificity for: Lap2, linked to cardiomyopathy; Nesprin 2, linked to Emery-Dreifuss muscular dystrophy and Lmo7, that regulates the Emery-Dreifuss muscular dystrophy linked emerin gene. Interestingly, the muscle-specific Lmo7 exon is rich in serine phosphorylation motifs, suggesting regulatory function. Muscle-specific splice variants in non-nuclear envelope proteins linked to other muscular dystrophies were also found. Nucleoporins tissue-specific variants were found for Nup54, Nup133, Nup153 and Nup358/RanBP2. RT-PCR confirmed novel Lmo7 and RanBP2 variants and specific knockdown of the Lmo7 variantreduced myogenic index. Nuclear envelope proteins were enriched for tissue-specific splice variants compared to the rest of the genome, suggesting that splice variants contribute to its tissue-specific functions.
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spelling pubmed-70001472020-02-19 Analysis of RNA-Seq datasets reveals enrichment of tissue-specific splice variants for nuclear envelope proteins Capitanchik, Charlotte Dixon, Charles R. Swanson, Selene K. Florens, Laurence Kerr, Alastair R. W. Schirmer, Eric C. Nucleus Special Issue on Laminopathies Laminopathies yield tissue-specific pathologies, yet arise from mutation of ubiquitously-expressed genes. A little investigated hypothesis to explain this is that the mutated proteins or their partners have tissue-specific splice variants. To test this, we analyzed RNA-Seq datasets, finding novel isoforms or isoform tissue-specificity for: Lap2, linked to cardiomyopathy; Nesprin 2, linked to Emery-Dreifuss muscular dystrophy and Lmo7, that regulates the Emery-Dreifuss muscular dystrophy linked emerin gene. Interestingly, the muscle-specific Lmo7 exon is rich in serine phosphorylation motifs, suggesting regulatory function. Muscle-specific splice variants in non-nuclear envelope proteins linked to other muscular dystrophies were also found. Nucleoporins tissue-specific variants were found for Nup54, Nup133, Nup153 and Nup358/RanBP2. RT-PCR confirmed novel Lmo7 and RanBP2 variants and specific knockdown of the Lmo7 variantreduced myogenic index. Nuclear envelope proteins were enriched for tissue-specific splice variants compared to the rest of the genome, suggesting that splice variants contribute to its tissue-specific functions. Taylor & Francis 2018-10-06 /pmc/articles/PMC7000147/ /pubmed/29912636 http://dx.doi.org/10.1080/19491034.2018.1469351 Text en © 2018 The Author(s). Published by Informa UK Limited, trading as Taylor & Francis Group. http://creativecommons.org/licenses/by/4.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Special Issue on Laminopathies
Capitanchik, Charlotte
Dixon, Charles R.
Swanson, Selene K.
Florens, Laurence
Kerr, Alastair R. W.
Schirmer, Eric C.
Analysis of RNA-Seq datasets reveals enrichment of tissue-specific splice variants for nuclear envelope proteins
title Analysis of RNA-Seq datasets reveals enrichment of tissue-specific splice variants for nuclear envelope proteins
title_full Analysis of RNA-Seq datasets reveals enrichment of tissue-specific splice variants for nuclear envelope proteins
title_fullStr Analysis of RNA-Seq datasets reveals enrichment of tissue-specific splice variants for nuclear envelope proteins
title_full_unstemmed Analysis of RNA-Seq datasets reveals enrichment of tissue-specific splice variants for nuclear envelope proteins
title_short Analysis of RNA-Seq datasets reveals enrichment of tissue-specific splice variants for nuclear envelope proteins
title_sort analysis of rna-seq datasets reveals enrichment of tissue-specific splice variants for nuclear envelope proteins
topic Special Issue on Laminopathies
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7000147/
https://www.ncbi.nlm.nih.gov/pubmed/29912636
http://dx.doi.org/10.1080/19491034.2018.1469351
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