CEP350 is a centrosomal protein essential for centrosome organization and ciliogenesis. Mechanistically, CEP350 stabilizes procentriolar seeds containing SASS6 and CPAP to enable centriole growth 1. It anchors microtubules to centrosomes and maintains microtubule network integrity 234. CEP350 acts upstream in a hierarchical recruitment pathway with FOP and CEP19 at the distal centriole, directing ciliary vesicle docking and initiating intraflagellar transport complex entry into nascent cilia 45. CEP350 also recruits and stabilizes CEP78, which negatively regulates CP110 to promote ciliogenesis 6. Additionally, CEP350 functions as a centriolar partner of the phosphatase PPP2R3C, which counterbalances MAP3K1 kinase activity to maintain centrosome function 7. Functionally, CEP350 depletion reduces microtubule stability and impairs ciliary assembly 84. Disease relevance includes ciliopathies, microcephaly, and cancer—CEP350 mutations associate with congenital syndromes involving gonadal dysgenesis, and CEP350 functions as a tumor suppressor in BRAF-driven melanoma 79. CEP350 variants also associate with smoking behavior in genome-wide studies 10.