Conserved TCP domain of Sas-4/CPAP is essential for pericentriolar material tethering during centrosome biogenesis

Zheng, Xiangdong; Gooi, Li Ming; Wason, Arpit; Gabriel, Elke; Mehrjardi, Narges Zare; Yang, Qian; Zhang, Xingrun; Debec, Alain; Basiri, Marcus L.; Avidor‐Reiss, Tomer; Pozniakovsky, Andrei; Poser, Ina; Šarić, Tomo; Hyman, Anthony A.; Li, Haitao; Gopalakrishnan, Jay

doi:10.1073/pnas.1317535111

Cited by 62 publications

(74 citation statements)

References 38 publications

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“…Additionally, we observed defects in the symmetric division of NPCs at the VZ, where ZIKV replication occurs ( Figures 4F-4I). Similar alterations were reported for genetically caused microcephaly (Gabriel et al, 2016;Hussain et al, 2012;Kalay et al, 2011;Lancaster et al, 2013;Zheng et al, 2014).…”

Section: Discussionsupporting

confidence: 83%

“…Defective centrosomes because of poor recruitment of centrosomal proteins are a hallmark of prematurely differentiating NPCs in microcephaly (Gabriel et al, 2016;Lancaster et al, 2013;Zheng et al, 2014). Indeed, ZIKV infection reduces the centrosomal recruitment of the microcephaly-linked proteins Cep152, PCNT, and CPAP (Figures 3A-3D; Bond et al, 2005;Cizmecioglu et al, 2010;Guernsey et al, 2010;Rauch et al, 2008).…”

Section: Zikv Infection Perturbs Centrosomal Structures Of Npcsmentioning

confidence: 99%

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Recent Zika Virus Isolates Induce Premature Differentiation of Neural Progenitors in Human Brain Organoids

et al. 2017

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SUMMARYThe recent Zika virus (ZIKV) epidemic is associated with microcephaly in newborns. Although the connection between ZIKV and neurodevelopmental defects is widely recognized, the underlying mechanisms are poorly understood. Here we show that two recently isolated strains of ZIKV, an American strain from an infected fetal brain (FB-GWUH-2016) and a closely-related Asian strain (H/PF/2013), productively infect human iPSC-derived brain organoids. Both of these strains readily target to and replicate in proliferating ventricular zone (VZ) apical progenitors. The main phenotypic effect was premature differentiation of neural progenitors associated with centrosome perturbation, even during early stages of infection, leading to progenitor depletion, disruption of the VZ, impaired neurogenesis, and cortical thinning. The infection pattern and cellular outcome differ from those seen with the extensively passaged ZIKV strain MR766. The structural changes we see after infection with these more recently isolated viral strains closely resemble those seen in ZIKV-associated microcephaly.

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Section: Discussionsupporting

confidence: 83%

Section: Zikv Infection Perturbs Centrosomal Structures Of Npcsmentioning

confidence: 99%

Recent Zika Virus Isolates Induce Premature Differentiation of Neural Progenitors in Human Brain Organoids

et al. 2017

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“…Asynchronously grown fibroblasts were pelleted, fixed with 2% glutaraldehyde (Electron Microscopy Sciences), and processed for electron microscopy as previously described (Gopalakrishnan et al , 2010; Zheng et al , 2014). The embedded cell pellets were ultra‐thin‐sectioned (80 nm), counterstained, and visualized for ultra‐structural details of centrosomes using a Zeiss 10A electron microscope.…”

Section: Methodsmentioning

confidence: 99%

CPAP promotes timely cilium disassembly to maintain neural progenitor pool

et al. 2016

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A mutation in the centrosomal‐P4.1‐associated protein (CPAP) causes Seckel syndrome with microcephaly, which is suggested to arise from a decline in neural progenitor cells (NPCs) during development. However, mechanisms of NPCs maintenance remain unclear. Here, we report an unexpected role for the cilium in NPCs maintenance and identify CPAP as a negative regulator of ciliary length independent of its role in centrosome biogenesis. At the onset of cilium disassembly, CPAP provides a scaffold for the cilium disassembly complex (CDC), which includes Nde1, Aurora A, and OFD1, recruited to the ciliary base for timely cilium disassembly. In contrast, mutated CPAP fails to localize at the ciliary base associated with inefficient CDC recruitment, long cilia, retarded cilium disassembly, and delayed cell cycle re‐entry leading to premature differentiation of patient iPS‐derived NPCs. Aberrant CDC function also promotes premature differentiation of NPCs in Seckel iPS‐derived organoids. Thus, our results suggest a role for cilia in microcephaly and its involvement during neurogenesis and brain size control.

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“…During centriole elongation CPAP then regulates the growth of centriolar microtubules 37–39,52 , which are inserted underneath a cap of CP110 36 . Interestingly, CPAP also interacts with STIL and it will be important to understand how CPAP and STIL modulate each other’s activities 32,111–113 .…”

Section: Control Of Centriole Numbermentioning

confidence: 99%

Once and only once: mechanisms of centriole duplication and their deregulation in disease

Nigg

Holland

2018

Nat Rev Mol Cell Biol

405

546

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Centrioles are conserved microtubule-based organelles that form the core of the centrosome and act as templates for the formation of cilia and flagella. Centrioles have important roles in most microtubule related processes, including motility, cell division and cell signaling. To coordinate these diverse cellular processes, centriole number must be tightly controlled. In cycling cells, one new centriole is formed next to each preexisting centriole in every cell cycle. Advances in imaging, proteomics, structural biology and genome editing have revealed new insights into centriole biogenesis, how centriole numbers are controlled and how alterations in these structures contribute to diseases such as cancer and neurodevelopmental disorders. Moreover, recent work has uncovered the existence of surveillance pathways that limit proliferation of cells with numerical centriole aberrations. Here we discuss recent progress in this field with a focus on signaling pathways and molecular mechanisms.

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Conserved TCP domain of Sas-4/CPAP is essential for pericentriolar material tethering during centrosome biogenesis

Cited by 62 publications

References 38 publications

Recent Zika Virus Isolates Induce Premature Differentiation of Neural Progenitors in Human Brain Organoids

Recent Zika Virus Isolates Induce Premature Differentiation of Neural Progenitors in Human Brain Organoids

CPAP promotes timely cilium disassembly to maintain neural progenitor pool

Once and only once: mechanisms of centriole duplication and their deregulation in disease

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