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Meis2 is essential for cranial and cardiac neural crest development

BACKGROUND: TALE-class homeodomain transcription factors Meis and Pbx play important roles in formation of the embryonic brain, eye, heart, cartilage or hematopoiesis. Loss-of-function studies of Pbx1, 2 and 3 and Meis1 documented specific functions in embryogenesis, however, functional studies of M...

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Autores principales: Machon, Ondrej, Masek, Jan, Machonova, Olga, Krauss, Stefan, Kozmik, Zbynek
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2015
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4636814/
https://www.ncbi.nlm.nih.gov/pubmed/26545946
http://dx.doi.org/10.1186/s12861-015-0093-6
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author Machon, Ondrej
Masek, Jan
Machonova, Olga
Krauss, Stefan
Kozmik, Zbynek
author_facet Machon, Ondrej
Masek, Jan
Machonova, Olga
Krauss, Stefan
Kozmik, Zbynek
author_sort Machon, Ondrej
collection PubMed
description BACKGROUND: TALE-class homeodomain transcription factors Meis and Pbx play important roles in formation of the embryonic brain, eye, heart, cartilage or hematopoiesis. Loss-of-function studies of Pbx1, 2 and 3 and Meis1 documented specific functions in embryogenesis, however, functional studies of Meis2 in mouse are still missing. We have generated a conditional allele of Meis2 in mice and shown that systemic inactivation of the Meis2 gene results in lethality by the embryonic day 14 that is accompanied with hemorrhaging. RESULTS: We show that neural crest cells express Meis2 and Meis2-defficient embryos display defects in tissues that are derived from the neural crest, such as an abnormal heart outflow tract with the persistent truncus arteriosus and abnormal cranial nerves. The importance of Meis2 for neural crest cells is further confirmed by means of conditional inactivation of Meis2 using crest-specific AP2α-IRES-Cre mouse. Conditional mutants display perturbed development of the craniofacial skeleton with severe anomalies in cranial bones and cartilages, heart and cranial nerve abnormalities. CONCLUSIONS: Meis2-null mice are embryonic lethal. Our results reveal a critical role of Meis2 during cranial and cardiac neural crest cells development in mouse. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (doi:10.1186/s12861-015-0093-6) contains supplementary material, which is available to authorized users.
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spelling pubmed-46368142015-11-08 Meis2 is essential for cranial and cardiac neural crest development Machon, Ondrej Masek, Jan Machonova, Olga Krauss, Stefan Kozmik, Zbynek BMC Dev Biol Research Article BACKGROUND: TALE-class homeodomain transcription factors Meis and Pbx play important roles in formation of the embryonic brain, eye, heart, cartilage or hematopoiesis. Loss-of-function studies of Pbx1, 2 and 3 and Meis1 documented specific functions in embryogenesis, however, functional studies of Meis2 in mouse are still missing. We have generated a conditional allele of Meis2 in mice and shown that systemic inactivation of the Meis2 gene results in lethality by the embryonic day 14 that is accompanied with hemorrhaging. RESULTS: We show that neural crest cells express Meis2 and Meis2-defficient embryos display defects in tissues that are derived from the neural crest, such as an abnormal heart outflow tract with the persistent truncus arteriosus and abnormal cranial nerves. The importance of Meis2 for neural crest cells is further confirmed by means of conditional inactivation of Meis2 using crest-specific AP2α-IRES-Cre mouse. Conditional mutants display perturbed development of the craniofacial skeleton with severe anomalies in cranial bones and cartilages, heart and cranial nerve abnormalities. CONCLUSIONS: Meis2-null mice are embryonic lethal. Our results reveal a critical role of Meis2 during cranial and cardiac neural crest cells development in mouse. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (doi:10.1186/s12861-015-0093-6) contains supplementary material, which is available to authorized users. BioMed Central 2015-11-06 /pmc/articles/PMC4636814/ /pubmed/26545946 http://dx.doi.org/10.1186/s12861-015-0093-6 Text en © Machon et al. 2015 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Research Article
Machon, Ondrej
Masek, Jan
Machonova, Olga
Krauss, Stefan
Kozmik, Zbynek
Meis2 is essential for cranial and cardiac neural crest development
title Meis2 is essential for cranial and cardiac neural crest development
title_full Meis2 is essential for cranial and cardiac neural crest development
title_fullStr Meis2 is essential for cranial and cardiac neural crest development
title_full_unstemmed Meis2 is essential for cranial and cardiac neural crest development
title_short Meis2 is essential for cranial and cardiac neural crest development
title_sort meis2 is essential for cranial and cardiac neural crest development
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4636814/
https://www.ncbi.nlm.nih.gov/pubmed/26545946
http://dx.doi.org/10.1186/s12861-015-0093-6
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