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

scientific article published on 13 June 2019

Modeling microcephaly with cerebral organoids reveals a WDR62-CEP170-KIF2A pathway promoting cilium disassembly in neural progenitors is …
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scholarly articleQ13442814

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P819ADS bibcode2019NatCo..10.2612Z
P356DOI10.1038/S41467-019-10497-2
P932PMC publication ID6565620
P698PubMed publication ID31197141

P50authorQiang ShaoQ47696131
Aimin LiuQ57569813
Bruce E HerringQ89025870
Yanhong ShiQ90740449
John L CollarQ92733481
P2093author name stringWei Zhang
Jian Xu
Jian-Fu Chen
Mei Yang
Shilpa Buch
Hui Lu
Zhen Zhao
Zhipeng Lu
Ming-Lei Guo
Stephanie Herrlinger
Si-Lu Yang
Edgar Fierro
P2860cites workGenome engineering using the CRISPR-Cas9 systemQ22122027
Evolution of the brain and intelligenceQ22337311
Brain-Region-Specific Organoids Using Mini-bioreactors for Modeling ZIKV ExposureQ24004552
Whole-exome sequencing identifies recessive WDR62 mutations in severe brain malformationsQ24296941
Cep164, a novel centriole appendage protein required for primary cilium formationQ24298367
Mutations in WDR62, encoding a centrosome-associated protein, cause microcephaly with simplified gyri and abnormal cortical architectureQ24301645
WDR62 is associated with the spindle pole and is mutated in human microcephalyQ24301673
The forkhead-associated domain protein Cep170 interacts with Polo-like kinase 1 and serves as a marker for mature centriolesQ24556578
Characterization of the intraflagellar transport complex B core: direct interaction of the IFT81 and IFT74/72 subunitsQ27919676
Cilium assembly and disassemblyQ28067455
Centriolar satellites assemble centrosomal microcephaly proteins to recruit CDK2 and promote centriole duplicationQ28115110
CDK5RAP2 regulates centriole engagement and cohesion in miceQ28505755
MCPH1 regulates the neuroprogenitor division mode by coupling the centrosomal cycle with mitotic entry through the Chk1-Cdc25 pathwayQ28506694
Chlamydomonas IFT88 and its mouse homologue, polycystic kidney disease gene tg737, are required for assembly of cilia and flagellaQ28593253
Human iPSC-Derived Cerebral Organoids Model Cellular Features of Lissencephaly and Reveal Prolonged Mitosis of Outer Radial GliaQ28595116
CPAP promotes timely cilium disassembly to maintain neural progenitor poolQ28973602
Cerebral organoids model human brain development and microcephalyQ28973619
The primary cilium: a signalling centre during vertebrate developmentQ29547197
The primary cilium as the cell's antenna: signaling at a sensory organelleQ29615165
Centrioles, centrosomes, and cilia in health and diseaseQ29615173
Neurogenic radial glia in the outer subventricular zone of human neocortexQ29616182
WD40 proteins propel cellular networks.Q30156873
Spindle poisons and cell fate: a tale of two pathways.Q30431071
Primary microcephaly: do all roads lead to Rome?Q33629131
Glial-Specific Functions of Microcephaly Protein WDR62 and Interaction with the Mitotic Kinase AURKA Are Essential for Drosophila Brain Growth.Q33906455
Developmental genetics of vertebrate glial-cell specification.Q34149150
Molecular genetic determinants of human brain sizeQ34529143
A genomic toolkit to investigate kinesin and myosin motor function in cellsQ34589246
Nde1-mediated inhibition of ciliogenesis affects cell cycle re-entryQ34796294
What disorders of cortical development tell us about the cortex: one plus one does not always make twoQ35115582
G1 phase regulation, area-specific cell cycle control, and cytoarchitectonics in the primate cortexQ35839746
Cdk5rap2 exposes the centrosomal root of microcephaly syndromesQ36021561
Mutations in TUBG1, DYNC1H1, KIF5C and KIF2A cause malformations of cortical development and microcephalyQ37304298
Self-organization of axial polarity, inside-out layer pattern, and species-specific progenitor dynamics in human ES cell-derived neocortexQ37395157
The Microtubule-Depolymerizing Activity of a Mitotic Kinesin Protein KIF2A Drives Primary Cilia Disassembly Coupled with Cell Proliferation.Q37398735
Disruptions in asymmetric centrosome inheritance and WDR62-Aurora kinase B interactions in primary microcephalyQ37685924
The primary cilium as a cellular receiver: organizing ciliary GPCR signalingQ38754528
Ciliary transition zone activation of phosphorylated Tctex-1 controls ciliary resorption, S-phase entry and fate of neural progenitorsQ39537369
A new subtype of progenitor cell in the mouse embryonic neocortexQ41917661
PLK1-mediated phosphorylation of WDR62/MCPH2 ensures proper mitotic spindle orientationQ41918159
Microcephaly Proteins Wdr62 and Aspm Define a Mother Centriole Complex Regulating Centriole Biogenesis, Apical Complex, and Cell FateQ41921410
The Microcephaly-Associated Protein Wdr62/CG7337 Is Required to Maintain Centrosome Asymmetry in Drosophila NeuroblastsQ41926666
Microcephaly disease gene Wdr62 regulates mitotic progression of embryonic neural stem cells and brain sizeQ41930126
Cdk4/cyclinD1 overexpression in neural stem cells shortens G1, delays neurogenesis, and promotes the generation and expansion of basal progenitors.Q42459454
OSVZ progenitors of human and ferret neocortex are epithelial-like and expand by integrin signalingQ42470977
Cell diversity and network dynamics in photosensitive human brain organoids.Q42615577
Ciliogenesis and cell cycle alterations contribute to KIF2A-related malformations of cortical development.Q47595186
Mitotic catastrophe: a mechanism for avoiding genomic instability.Q52610466
Do cilia put brakes on the cell cycle?Q83771992
Microtubule-depolymerizing kinesinsQ87130000
P433issue1
P407language of work or nameEnglishQ1860
P921main subjectorganoidQ11293125
microcephalyQ431643
P304page(s)2612
P577publication date2019-06-13
P1433published inNature CommunicationsQ573880
P1476titleModeling microcephaly with cerebral organoids reveals a WDR62-CEP170-KIF2A pathway promoting cilium disassembly in neural progenitors
P478volume10

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cites work (P2860)
Q97516713Application of Fused Organoid Models to Study Human Brain Development and Neural Disorders
Q96128071Brain organoids: an ensemble of bioassays to investigate human neurodevelopment and disease
Q89960154Cerebral organoid and mouse models reveal a RAB39b-PI3K-mTOR pathway-dependent dysregulation of cortical development leading to macrocephaly/autism phenotypes
Q84957742Cortical Malformations: Lessons in Human Brain Development
Q90064979FLS2 is a CDK-like kinase that directly binds IFT70 and is required for proper ciliary disassembly in Chlamydomonas
Q95840623Human Brain Organoids to Decode Mechanisms of Microcephaly
Q102075330RRP7A links primary microcephaly to dysfunction of ribosome biogenesis, resorption of primary cilia, and neurogenesis
Q96304780Urine Sample-Derived Cerebral Organoids Suitable for Studying Neurodevelopment and Pharmacological Responses

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