CEP63 is a centrosomal scaffolding protein essential for centriole duplication and spindle assembly. CEP63 functions as a key component of the pericentriolar matrix, interacting with CEP152, CEP57, and WDR62 to form a stepwise assembled complex that recruits CDK2 for centriole duplication 12. CEP63 also recruits CDK1 to centrosomes 3 and maintains proper spatial organization of centrosomal proteins, including anchoring CEP152 in ninefold symmetry during centriole maturation 4. Beyond cell cycle regulation, CEP63 plays a critical role in DNA damage response; following double-strand breaks, it is removed from centrosomes, leading to spindle assembly inactivation and mitotic delay 3. Recently identified functions include stabilization of the RNA-binding protein FXR1 through inhibition of its K63-linked ubiquitination 5, and regulation of innate immunity by modulating TBK1 centrosomal localization 6. Loss-of-function mutations in CEP63 cause Seckel syndrome 6, characterized by microcephaly, growth restriction, and intellectual disability 78, with disease pathology arising from centrosome-based mitotic errors and p53-dependent neural progenitor cell death rather than aberrant DNA damage response 8.