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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Main Authors:	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
Format:	Article
Language:	English
Published:	Nature Publishing Group 2019-06-01
Series:	Nature Communications
Online Access:	https://doi.org/10.1038/s41467-019-10497-2

id	doaj-fe809d4eaad64cd5995ce2a720fd1c05
record_format	Article
spelling	doaj-fe809d4eaad64cd5995ce2a720fd1c052021-05-11T12:06:20ZengNature Publishing GroupNature Communications2041-17232019-06-0110111410.1038/s41467-019-10497-2Modeling microcephaly with cerebral organoids reveals a WDR62–CEP170–KIF2A pathway promoting cilium disassembly in neural progenitorsWei Zhang0Si-Lu Yang1Mei Yang2Stephanie Herrlinger3Qiang Shao4John L. Collar5Edgar Fierro6Yanhong Shi7Aimin Liu8Hui Lu9Bruce E. Herring10Ming-Lei Guo11Shilpa Buch12Zhen Zhao13Jian Xu14Zhipeng Lu15Jian-Fu Chen16Center for Craniofacial Molecular Biology, University of Southern California (USC)Department of Genetics, University of GeorgiaCenter for Craniofacial Molecular Biology, University of Southern California (USC)Department of Genetics, University of GeorgiaCenter for Craniofacial Molecular Biology, University of Southern California (USC)Department of Genetics, University of GeorgiaDepartment of Genetics, University of GeorgiaDivision of Stem Cell Biology Research, Department of Developmental and Stem Cell Biology, Beckman Research Institute of City of HopeDepartment of Biology, Eberly College of Science, The Pennsylvania State UniversityDepartment of Pharmacology and Physiology, The George Washington UniversityDepartment of Biological Sciences, University of Southern CaliforniaDepartment of Pharmacology and Experimental Neuroscience, University of Nebraska Medical CenterDepartment of Pharmacology and Experimental Neuroscience, University of Nebraska Medical CenterZilkha Neurogenetic Institute, Keck School of Medicine, University of Southern CaliforniaCenter for Craniofacial Molecular Biology, University of Southern California (USC)Department of Pharmacology and Pharmaceutical Sciences, University of Southern CaliforniaCenter for Craniofacial Molecular Biology, University of Southern California (USC)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.https://doi.org/10.1038/s41467-019-10497-2
collection	DOAJ
language	English
format	Article
sources	DOAJ
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
spellingShingle	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 Nature Communications
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 Publishing Group
series	Nature Communications
issn	2041-1723
publishDate	2019-06-01
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.
url	https://doi.org/10.1038/s41467-019-10497-2
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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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