Itm2a is a Pax3 target gene, expressed at sites of skeletal muscle formation in vivo.

The paired-box homeodomain transcription factor Pax3 is a key regulator of the nervous system, neural crest and skeletal muscle development. Despite the important role of this transcription factor, very few direct target genes have been characterized. We show that Itm2a, which encodes a type 2 trans...

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Main Authors: Mounia Lagha, Alicia Mayeuf-Louchart, Ted Chang, Didier Montarras, Didier Rocancourt, Antoine Zalc, Jay Kormish, Kenneth S Zaret, Margaret E Buckingham, Frederic Relaix
Format: Article
Language:English
Published: Public Library of Science (PLoS) 2013-01-01
Series:PLoS ONE
Online Access:http://europepmc.org/articles/PMC3641095?pdf=render
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author Mounia Lagha
Alicia Mayeuf-Louchart
Ted Chang
Didier Montarras
Didier Rocancourt
Antoine Zalc
Jay Kormish
Kenneth S Zaret
Margaret E Buckingham
Frederic Relaix
author_facet Mounia Lagha
Alicia Mayeuf-Louchart
Ted Chang
Didier Montarras
Didier Rocancourt
Antoine Zalc
Jay Kormish
Kenneth S Zaret
Margaret E Buckingham
Frederic Relaix
author_sort Mounia Lagha
collection DOAJ
description The paired-box homeodomain transcription factor Pax3 is a key regulator of the nervous system, neural crest and skeletal muscle development. Despite the important role of this transcription factor, very few direct target genes have been characterized. We show that Itm2a, which encodes a type 2 transmembrane protein, is a direct Pax3 target in vivo, by combining genetic approaches and in vivo chromatin immunoprecipitation assays. We have generated a conditional mutant allele for Itm2a, which is an imprinted gene, by flanking exons 2-4 with loxP sites and inserting an IRESnLacZ reporter in the 3' UTR of the gene. The LacZ reporter reproduces the expression profile of Itm2a, and allowed us to further characterize its expression at sites of myogenesis, in the dermomyotome and myotome of somites, and in limb buds, in the mouse embryo. We further show that Itm2a is not only expressed in adult muscle fibres but also in the satellite cells responsible for regeneration. Itm2a mutant mice are viable and fertile with no overt phenotype during skeletal muscle formation or regeneration. Potential compensatory mechanisms are discussed.
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spelling doaj.art-6f367ee861654522a5bfbfbd3c2986b42022-12-21T23:55:06ZengPublic Library of Science (PLoS)PLoS ONE1932-62032013-01-0185e6314310.1371/journal.pone.0063143Itm2a is a Pax3 target gene, expressed at sites of skeletal muscle formation in vivo.Mounia LaghaAlicia Mayeuf-LouchartTed ChangDidier MontarrasDidier RocancourtAntoine ZalcJay KormishKenneth S ZaretMargaret E BuckinghamFrederic RelaixThe paired-box homeodomain transcription factor Pax3 is a key regulator of the nervous system, neural crest and skeletal muscle development. Despite the important role of this transcription factor, very few direct target genes have been characterized. We show that Itm2a, which encodes a type 2 transmembrane protein, is a direct Pax3 target in vivo, by combining genetic approaches and in vivo chromatin immunoprecipitation assays. We have generated a conditional mutant allele for Itm2a, which is an imprinted gene, by flanking exons 2-4 with loxP sites and inserting an IRESnLacZ reporter in the 3' UTR of the gene. The LacZ reporter reproduces the expression profile of Itm2a, and allowed us to further characterize its expression at sites of myogenesis, in the dermomyotome and myotome of somites, and in limb buds, in the mouse embryo. We further show that Itm2a is not only expressed in adult muscle fibres but also in the satellite cells responsible for regeneration. Itm2a mutant mice are viable and fertile with no overt phenotype during skeletal muscle formation or regeneration. Potential compensatory mechanisms are discussed.http://europepmc.org/articles/PMC3641095?pdf=render
spellingShingle Mounia Lagha
Alicia Mayeuf-Louchart
Ted Chang
Didier Montarras
Didier Rocancourt
Antoine Zalc
Jay Kormish
Kenneth S Zaret
Margaret E Buckingham
Frederic Relaix
Itm2a is a Pax3 target gene, expressed at sites of skeletal muscle formation in vivo.
PLoS ONE
title Itm2a is a Pax3 target gene, expressed at sites of skeletal muscle formation in vivo.
title_full Itm2a is a Pax3 target gene, expressed at sites of skeletal muscle formation in vivo.
title_fullStr Itm2a is a Pax3 target gene, expressed at sites of skeletal muscle formation in vivo.
title_full_unstemmed Itm2a is a Pax3 target gene, expressed at sites of skeletal muscle formation in vivo.
title_short Itm2a is a Pax3 target gene, expressed at sites of skeletal muscle formation in vivo.
title_sort itm2a is a pax3 target gene expressed at sites of skeletal muscle formation in vivo
url http://europepmc.org/articles/PMC3641095?pdf=render
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