CEP41 (centrosomal protein 41) is a centrosomal and ciliary protein with multifaceted roles in ciliary and cell cycle biology. Primarily, CEP41 regulates tubulin glutamylation, a critical post-translational modification of ciliary axonemal tubulin 1. CEP41 localizes to basal bodies and primary cilia, where it facilitates ciliary entry of TTLL6, a polyglutamylase enzyme essential for proper glutamylation 1. Beyond ciliary function, CEP41 acts as a microtubule-associated protein with microtubule-stabilizing activity; it binds preformed microtubules, promotes nucleation, and suppresses disassembly through its conserved rhodanese homology domain 2. CEP41 depletion disrupts interphase microtubule networks and delays cell cycle progression, suggesting roles in cell proliferation regulation 2. In endothelial cells, CEP41-mediated tubulin glutamylation drives angiogenesis through AURKA-dependent deciliation and HIF1α-VEGF pathway activation 3. Clinically, homozygous CEP41 mutations cause Joubert syndrome 15, a neurodevelopmental ciliopathy featuring hindbrain malformations 14. Heterozygous CEP41 missense variants are associated with autism spectrum disorder, affecting axonal tract development and neural crest migration 5. Additionally, CEP41 hypermethylation correlates with male infertility phenotypes 6.