Cep295 is a conserved scaffold protein required for generation of a bona fide mother centriole

Tsuchiya, Yuki; Yoshiba, Satoko; Gupta, Akshari; Watanabe, Kageaki; Kitagawa, Daiju

doi:10.1038/ncomms12567

Cited by 61 publications

(76 citation statements)

References 49 publications

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“…Importantly, recruitment of Asl in Drosophila embryos only occurs after disengagement, indicating that these licensing processes occur sequentially 93 . In mammalian cells, CEP295 directly binds to CEP192 and contributes to the stabilization of centrioles after the loss of the cartwheel upon mitotic exit 89,94 . Considering that CEP152 and CEP192 form scaffolds for the recruitment of PLK4 95–99 , the kinase essential for centriole duplication (see below), these results explain why post-mitotic PCM assembly is required to confer duplication competence to procentrioles 91 .…”

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

420

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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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

420

546

View full text Add to dashboard Cite

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“…Therefore, to distinguish whether these excess Cep192 dots reflect centrosome amplification, centriole disengagement, or PCM fragmentation, we next performed immunofluorescence analyses using specific antibodies against centriolar glutamylated microtubules (GT335) and Cep152, a mother centriole marker, in lymphocyte cells (Tsuchiya et al. ). We found that, in the CHAMP1 ‐mutant patient's cells, all Cep192 foci on the multipolar spindle poles contained GT335 dots (Fig.…”

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confidence: 99%

Disturbed chromosome segregation and multipolar spindle formation in a patient with CHAMP 1 mutation

Okamoto

Tsuchiya

Kuki

et al. 2017

Mol Genet Genomic Med

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BackgroundPatients with intellectual disability (ID) typically exhibit significant defects in both intelligence and adaptive behavior. Aberration of several genes involved in proper progression of mitosis has been reported to underlie ID. Here, we report a new patient with a novel mutation of CHAMP1.MethodsWhole exome sequencing (WES) analysis was performed. We isolated lymphoblast cells from the CHAMP1 patient and observed chromosome segregation.ResultsWe identified a de novo frameshift mutation in CHAMP1. We find that these cells exhibit an increase in centrosome number and resulting multipolar spindle formation. The phenotypes observed in the patient's lymphoblastoid cells were presumably because of cytokinesis failure. We also confirm the identical phenotypes in human culture cells depleted of CHAMP1.Conclusion CHAMP1 encodes a protein regulating kinetochore–microtubule attachment and chromosome segregation. These data strongly support that CHAMP1 mutations cause ID, and suggest that CHAMP1 is critical for progression of cytokinesis and maintenance of centrosome number.

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“…Recently, Cep295 is identified as the key protein for the conversion of daughter centrioles into functional mother centrioles, and the loading of Cep152 by Cep135 and Cep295 to daughter centrioles is critical for the conversion (Fu et al, 2016; Tsuchiya et al, 2016). Interestingly, Cep295 is only required for targeting of Cep152 to the young mother centriole, but not to the old mother centriole, implying that other scaffold proteins might play a role in anchoring Cep152 to the old mother centriole.…”

Section: Resultsmentioning

confidence: 99%

Cep57 and Cep57l1 cooperate to recruit the Cep63-Cep152 complex for centriole biogenesis

Zhao

Yang

Duan

et al. 2019

Preprint

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Cep152 and Cep63 act as the cradle and recruit Plk4 to initiate the centriole biogenesis in a mother centriole dependent manner. However, how the Cep152-Cep63 complex is targeted to the proximal end of mother centrioles is unclear. In this study, we show that Cep57 and its paralog, Cep57l1, colocalize with Cep63 and Cep152 at the proximal end of mother centrioles in both cycling cells and differentiated multiciliated cells. Both Cep57 and Cep57l1 associate with the Cep152-Cep63 complex by directly binding to the centrosomal targeting region of Cep63. Co-depletion of Cep57 and Cep57l1 blocks the loading of Cep63-Cep152 to the centriole and subsequently prevents centriole duplication. We propose that Cep57 and Cep57l1 act together to ensure the recruitment of the Cep63-Cep152 complex to the mother centrioles for procentriole formation. Summary statement Cep57 and its paralog, Cep57l1, cooperatively act as a molecular scaffold to recruit the Cep63-Cep152 cradle to the proximal end of centrioles in the early stages of centriole duplication.

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Cep295 is a conserved scaffold protein required for generation of a bona fide mother centriole

Cited by 61 publications

References 49 publications

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

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

Disturbed chromosome segregation and multipolar spindle formation in a patient with CHAMP 1 mutation

Cep57 and Cep57l1 cooperate to recruit the Cep63-Cep152 complex for centriole biogenesis

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