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...
| Main Authors: | , , , , , , , , , , , , , , , , |
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| Format: | Article |
| Language: | English |
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Nature Portfolio
2019-06-01
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| Series: | Nature Communications |
| Online Access: | https://doi.org/10.1038/s41467-019-10497-2 |
| _version_ | 1830416997582635008 |
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| 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 |
| collection | DOAJ |
| 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. |
| first_indexed | 2024-12-20T21:34:18Z |
| format | Article |
| id | doaj.art-fe809d4eaad64cd5995ce2a720fd1c05 |
| institution | Directory Open Access Journal |
| issn | 2041-1723 |
| language | English |
| last_indexed | 2024-12-20T21:34:18Z |
| publishDate | 2019-06-01 |
| publisher | Nature Portfolio |
| record_format | Article |
| series | Nature Communications |
| spelling | doaj.art-fe809d4eaad64cd5995ce2a720fd1c052022-12-21T19:25:59ZengNature PortfolioNature 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 |
| 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 |
| title | 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_short | 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 |
| url | https://doi.org/10.1038/s41467-019-10497-2 |
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