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In brain development, Wdr62 and Aspm localize to the proximal end of the mother centriole and interact physically, with Wdr62 required for Aspm localization, and both proteins, as well as microcephaly protein Cep63, required to localize CENPJ/CPAP/Sas-4, a final common target.
Cep63-deficient mice recapitulate Seckel syndrome pathology, microcephaly and dwarfism. Cep63 loss severely impairs meiotic recombination, leading to profound male infertility.
both mouse and human Cep63 and Cep152 cooperate to ensure efficient centriole duplication by promoting the accumulation of essential centriole duplication factors upstream of SAS-6 recruitment and procentriole formation.
Cep63 regulates mother-centriole-dependent centriole duplication.
Study shows that Cep63 and Cep152 cooperatively generate a heterotetrameric alpha-helical bundle that functions in conjunction with its neighboring hydrophobic motifs to self-assemble into a higher-order cylindrical architecture capable of recruiting downstream components, including Plk4, a key regulator for centriole duplication. Mutations disrupting the self-assembly abrogate Plk4-mediated centriole duplication.
CEP63 is particularly important for brain development and might control the proliferation and migration of cells when those two events need to be highly coordinated.
centrosomal Cdk1 signals were strongly diminished in cells treated with Cep63 siRNA when sc-54 was used for Cdk1 labeling
Cep57, Cep63, and Cep152 are parts of a ring-like complex localizing around the proximal end of centrioles.
One new SNP (rs112926188) in CEP63O was reported. Analysis showed that rs112926188 allele substituting proline at the 61st residue position of CEP63 produced more flexibility in three-deimensional space.
Findings define Cep63 as a centrosomal recruitment factor for Cdk1 that is essential for mitotic entry, providing a physical link between the centrosome and the cell-cycle machinery.
CEP63 transcripts exhibited three different expression patterns, downregulation was found in about 50% of the cases analyzed and CEP63 gene transcription was associated with invasive tumors.
ATM and ATR control mitotic events in vertebrate cells by targeting CEP63 and centrosome dependent spindle assembly.
This gene encodes a protein with six coiled-coil domains. The protein is localized to the centrosome, a non-membraneous organelle that functions as the major microtubule-organizing center in animal cells. Several alternatively spliced transcript variants have been found, but their biological validity has not been determined.
centrosomal protein 63kDa
, centrosomal protein 63
, centrosomal protein of 63 kDa-like
, centrosomal protein of 63 kDa
, centrosome protein Cep63
, centrosome protein CEP63
, Centrosomal protein of 63 kDa-A
, Centrosomal protein of 63 kDa-B