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A missense mutation in zbtb17 blocks the earliest steps of T cell differentiation in zebrafish

T cells are an evolutionarily conserved feature of the adaptive immune systems of vertebrates. Comparative studies using evolutionarily distant species hold great promise for unraveling the genetic landscape underlying this process. To this end, we used ENU mutagenesis to generate mutant zebrafish w...

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Autores principales: Lawir, Divine-Fondzenyuy, Iwanami, Norimasa, Schorpp, Michael, Boehm, Thomas
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5339814/
https://www.ncbi.nlm.nih.gov/pubmed/28266617
http://dx.doi.org/10.1038/srep44145
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author Lawir, Divine-Fondzenyuy
Iwanami, Norimasa
Schorpp, Michael
Boehm, Thomas
author_facet Lawir, Divine-Fondzenyuy
Iwanami, Norimasa
Schorpp, Michael
Boehm, Thomas
author_sort Lawir, Divine-Fondzenyuy
collection PubMed
description T cells are an evolutionarily conserved feature of the adaptive immune systems of vertebrates. Comparative studies using evolutionarily distant species hold great promise for unraveling the genetic landscape underlying this process. To this end, we used ENU mutagenesis to generate mutant zebrafish with specific aberrations in early T cell development. Here, we describe the identification of a recessive missense mutation in the transcriptional regulator zbtb17 (Q562K), which affects the ninth zinc finger module of the protein. Homozygous mutant fish exhibit an early block of intrathymic T cell development, as a result of impaired thymus colonization owing to reduced expression of the gene encoding the homing receptor ccr9a, and inefficient T cell differentiation owing to reduced expression of socs1a. Our results reveal the zbtb17-socs1 axis as an evolutionarily conserved central regulatory module of early T cell development of vertebrates.
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spelling pubmed-53398142017-03-10 A missense mutation in zbtb17 blocks the earliest steps of T cell differentiation in zebrafish Lawir, Divine-Fondzenyuy Iwanami, Norimasa Schorpp, Michael Boehm, Thomas Sci Rep Article T cells are an evolutionarily conserved feature of the adaptive immune systems of vertebrates. Comparative studies using evolutionarily distant species hold great promise for unraveling the genetic landscape underlying this process. To this end, we used ENU mutagenesis to generate mutant zebrafish with specific aberrations in early T cell development. Here, we describe the identification of a recessive missense mutation in the transcriptional regulator zbtb17 (Q562K), which affects the ninth zinc finger module of the protein. Homozygous mutant fish exhibit an early block of intrathymic T cell development, as a result of impaired thymus colonization owing to reduced expression of the gene encoding the homing receptor ccr9a, and inefficient T cell differentiation owing to reduced expression of socs1a. Our results reveal the zbtb17-socs1 axis as an evolutionarily conserved central regulatory module of early T cell development of vertebrates. Nature Publishing Group 2017-03-07 /pmc/articles/PMC5339814/ /pubmed/28266617 http://dx.doi.org/10.1038/srep44145 Text en Copyright © 2017, The Author(s) http://creativecommons.org/licenses/by/4.0/ This work is licensed under a Creative Commons Attribution 4.0 International License. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in the credit line; if the material is not included under the Creative Commons license, users will need to obtain permission from the license holder to reproduce the material. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/
spellingShingle Article
Lawir, Divine-Fondzenyuy
Iwanami, Norimasa
Schorpp, Michael
Boehm, Thomas
A missense mutation in zbtb17 blocks the earliest steps of T cell differentiation in zebrafish
title A missense mutation in zbtb17 blocks the earliest steps of T cell differentiation in zebrafish
title_full A missense mutation in zbtb17 blocks the earliest steps of T cell differentiation in zebrafish
title_fullStr A missense mutation in zbtb17 blocks the earliest steps of T cell differentiation in zebrafish
title_full_unstemmed A missense mutation in zbtb17 blocks the earliest steps of T cell differentiation in zebrafish
title_short A missense mutation in zbtb17 blocks the earliest steps of T cell differentiation in zebrafish
title_sort missense mutation in zbtb17 blocks the earliest steps of t cell differentiation in zebrafish
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5339814/
https://www.ncbi.nlm.nih.gov/pubmed/28266617
http://dx.doi.org/10.1038/srep44145
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