Cargando…

Deletion of the chd7 Hinders Oligodendrocyte Progenitor Cell Development and Myelination in Zebrafish

CHD7, an encoding ATP-dependent chromodomain helicase DNA-binding protein 7, has been identified as the causative gene involved in CHARGE syndrome (Coloboma of the eye, Heart defects, Atresia choanae, Retardation of growth and/or development, Genital abnormalities and Ear abnormalities). Although st...

Descripción completa

Detalles Bibliográficos
Autores principales: Shi, Lingyu, Wang, Zongyi, Li, Yujiao, Song, Zheng, Yin, Wu, Hu, Bing
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10488005/
https://www.ncbi.nlm.nih.gov/pubmed/37686337
http://dx.doi.org/10.3390/ijms241713535
_version_ 1785103376031154176
author Shi, Lingyu
Wang, Zongyi
Li, Yujiao
Song, Zheng
Yin, Wu
Hu, Bing
author_facet Shi, Lingyu
Wang, Zongyi
Li, Yujiao
Song, Zheng
Yin, Wu
Hu, Bing
author_sort Shi, Lingyu
collection PubMed
description CHD7, an encoding ATP-dependent chromodomain helicase DNA-binding protein 7, has been identified as the causative gene involved in CHARGE syndrome (Coloboma of the eye, Heart defects, Atresia choanae, Retardation of growth and/or development, Genital abnormalities and Ear abnormalities). Although studies in rodent models have expanded our understanding of CHD7, its role in oligodendrocyte (OL) differentiation and myelination in zebrafish is still unclear. In this study, we generated a chd7-knockout strain with CRISPR/Cas9 in zebrafish. We observed that knockout (KO) of chd7 intensely impeded the oligodendrocyte progenitor cells’ (OPCs) migration and myelin formation due to massive expression of chd7 in oilg2(+) cells, which might provoke upregulation of the MAPK signal pathway. Thus, our study demonstrates that chd7 is critical to oligodendrocyte migration and myelination during early development in zebrafish and describes a mechanism potentially associated with CHARGE syndrome.
format Online
Article
Text
id pubmed-10488005
institution National Center for Biotechnology Information
language English
publishDate 2023
publisher MDPI
record_format MEDLINE/PubMed
spelling pubmed-104880052023-09-09 Deletion of the chd7 Hinders Oligodendrocyte Progenitor Cell Development and Myelination in Zebrafish Shi, Lingyu Wang, Zongyi Li, Yujiao Song, Zheng Yin, Wu Hu, Bing Int J Mol Sci Article CHD7, an encoding ATP-dependent chromodomain helicase DNA-binding protein 7, has been identified as the causative gene involved in CHARGE syndrome (Coloboma of the eye, Heart defects, Atresia choanae, Retardation of growth and/or development, Genital abnormalities and Ear abnormalities). Although studies in rodent models have expanded our understanding of CHD7, its role in oligodendrocyte (OL) differentiation and myelination in zebrafish is still unclear. In this study, we generated a chd7-knockout strain with CRISPR/Cas9 in zebrafish. We observed that knockout (KO) of chd7 intensely impeded the oligodendrocyte progenitor cells’ (OPCs) migration and myelin formation due to massive expression of chd7 in oilg2(+) cells, which might provoke upregulation of the MAPK signal pathway. Thus, our study demonstrates that chd7 is critical to oligodendrocyte migration and myelination during early development in zebrafish and describes a mechanism potentially associated with CHARGE syndrome. MDPI 2023-08-31 /pmc/articles/PMC10488005/ /pubmed/37686337 http://dx.doi.org/10.3390/ijms241713535 Text en © 2023 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Shi, Lingyu
Wang, Zongyi
Li, Yujiao
Song, Zheng
Yin, Wu
Hu, Bing
Deletion of the chd7 Hinders Oligodendrocyte Progenitor Cell Development and Myelination in Zebrafish
title Deletion of the chd7 Hinders Oligodendrocyte Progenitor Cell Development and Myelination in Zebrafish
title_full Deletion of the chd7 Hinders Oligodendrocyte Progenitor Cell Development and Myelination in Zebrafish
title_fullStr Deletion of the chd7 Hinders Oligodendrocyte Progenitor Cell Development and Myelination in Zebrafish
title_full_unstemmed Deletion of the chd7 Hinders Oligodendrocyte Progenitor Cell Development and Myelination in Zebrafish
title_short Deletion of the chd7 Hinders Oligodendrocyte Progenitor Cell Development and Myelination in Zebrafish
title_sort deletion of the chd7 hinders oligodendrocyte progenitor cell development and myelination in zebrafish
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10488005/
https://www.ncbi.nlm.nih.gov/pubmed/37686337
http://dx.doi.org/10.3390/ijms241713535
work_keys_str_mv AT shilingyu deletionofthechd7hindersoligodendrocyteprogenitorcelldevelopmentandmyelinationinzebrafish
AT wangzongyi deletionofthechd7hindersoligodendrocyteprogenitorcelldevelopmentandmyelinationinzebrafish
AT liyujiao deletionofthechd7hindersoligodendrocyteprogenitorcelldevelopmentandmyelinationinzebrafish
AT songzheng deletionofthechd7hindersoligodendrocyteprogenitorcelldevelopmentandmyelinationinzebrafish
AT yinwu deletionofthechd7hindersoligodendrocyteprogenitorcelldevelopmentandmyelinationinzebrafish
AT hubing deletionofthechd7hindersoligodendrocyteprogenitorcelldevelopmentandmyelinationinzebrafish