Modeling microcephaly with cerebral organoids reveals a WDR62–CEP170–KIF2A pathway promoting cilium disassembly in neural progenitors

Mutations in WDR62 are the second most common genetic cause of autosomal recessive primary microcephaly, yet the molecular mechanisms underlying this pathogenesis remain unclear. Here, authors demonstrate that WDR62 depletion leads to neural precursor cell depletion and microcephaly via WDR62-CEP170...

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Autores principales:	Wei Zhang, Si-Lu Yang, Mei Yang, Stephanie Herrlinger, Qiang Shao, John L. Collar, Edgar Fierro, Yanhong Shi, Aimin Liu, Hui Lu, Bruce E. Herring, Ming-Lei Guo, Shilpa Buch, Zhen Zhao, Jian Xu, Zhipeng Lu, Jian-Fu Chen
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2019
Materias:	Science Q
Acceso en línea:	https://doaj.org/article/fe809d4eaad64cd5995ce2a720fd1c05
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spelling	oai:doaj.org-article:fe809d4eaad64cd5995ce2a720fd1c052021-12-02T16:57:33ZModeling microcephaly with cerebral organoids reveals a WDR62–CEP170–KIF2A pathway promoting cilium disassembly in neural progenitors10.1038/s41467-019-10497-22041-1723https://doaj.org/article/fe809d4eaad64cd5995ce2a720fd1c052019-06-01T00:00:00Zhttps://doi.org/10.1038/s41467-019-10497-2https://doaj.org/toc/2041-1723Mutations in WDR62 are the second most common genetic cause of autosomal recessive primary microcephaly, yet the molecular mechanisms underlying this pathogenesis remain unclear. Here, authors demonstrate that WDR62 depletion leads to neural precursor cell depletion and microcephaly via WDR62-CEP170-KIF2A pathway that promotes cilium disassembly.Wei ZhangSi-Lu YangMei YangStephanie HerrlingerQiang ShaoJohn L. CollarEdgar FierroYanhong ShiAimin LiuHui LuBruce E. HerringMing-Lei GuoShilpa BuchZhen ZhaoJian XuZhipeng LuJian-Fu ChenNature PortfolioarticleScienceQENNature Communications, Vol 10, Iss 1, Pp 1-14 (2019)
institution	DOAJ
collection	DOAJ
language	EN
topic	Science Q
spellingShingle	Science Q Wei Zhang Si-Lu Yang Mei Yang Stephanie Herrlinger Qiang Shao John L. Collar Edgar Fierro Yanhong Shi Aimin Liu Hui Lu Bruce E. Herring Ming-Lei Guo Shilpa Buch Zhen Zhao Jian Xu Zhipeng Lu Jian-Fu Chen Modeling microcephaly with cerebral organoids reveals a WDR62–CEP170–KIF2A pathway promoting cilium disassembly in neural progenitors
description	Mutations in WDR62 are the second most common genetic cause of autosomal recessive primary microcephaly, yet the molecular mechanisms underlying this pathogenesis remain unclear. Here, authors demonstrate that WDR62 depletion leads to neural precursor cell depletion and microcephaly via WDR62-CEP170-KIF2A pathway that promotes cilium disassembly.
format	article
author	Wei Zhang Si-Lu Yang Mei Yang Stephanie Herrlinger Qiang Shao John L. Collar Edgar Fierro Yanhong Shi Aimin Liu Hui Lu Bruce E. Herring Ming-Lei Guo Shilpa Buch Zhen Zhao Jian Xu Zhipeng Lu Jian-Fu Chen
author_facet	Wei Zhang Si-Lu Yang Mei Yang Stephanie Herrlinger Qiang Shao John L. Collar Edgar Fierro Yanhong Shi Aimin Liu Hui Lu Bruce E. Herring Ming-Lei Guo Shilpa Buch Zhen Zhao Jian Xu Zhipeng Lu Jian-Fu Chen
author_sort	Wei Zhang
title	Modeling microcephaly with cerebral organoids reveals a WDR62–CEP170–KIF2A pathway promoting cilium disassembly in neural progenitors
title_short	Modeling microcephaly with cerebral organoids reveals a WDR62–CEP170–KIF2A pathway promoting cilium disassembly in neural progenitors
title_full	Modeling microcephaly with cerebral organoids reveals a WDR62–CEP170–KIF2A pathway promoting cilium disassembly in neural progenitors
title_fullStr	Modeling microcephaly with cerebral organoids reveals a WDR62–CEP170–KIF2A pathway promoting cilium disassembly in neural progenitors
title_full_unstemmed	Modeling microcephaly with cerebral organoids reveals a WDR62–CEP170–KIF2A pathway promoting cilium disassembly in neural progenitors
title_sort	modeling microcephaly with cerebral organoids reveals a wdr62–cep170–kif2a pathway promoting cilium disassembly in neural progenitors
publisher	Nature Portfolio
publishDate	2019
url	https://doaj.org/article/fe809d4eaad64cd5995ce2a720fd1c05
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Modeling microcephaly with cerebral organoids reveals a WDR62–CEP170–KIF2A pathway promoting cilium disassembly in neural progenitors

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