CNTROB encodes centrobin, a centrosomal protein essential for centriole duplication and primary ciliogenesis in vertebrates. The protein primarily localizes to daughter centrioles and associates with mother centrioles upon serum starvation 1. Functionally, centrobin is required for proper centriole duplication, with its loss leading to increased frequencies of monocentriolar and acentriolar cells 1. The protein plays a critical role in primary ciliogenesis by facilitating axonemal extension after mother centriole docking, requiring its C-terminal portion that interacts with CP110 and tubulin 1. In spermatogenesis, centrobin localizes to the manchette, centrosome, and acroplaxome marginal ring, where it interacts with keratin 5-containing intermediate filaments and is essential for proper spermatid head shaping and head-tail coupling apparatus assembly 2. Mutations in CNTROB cause male infertility due to spermatid decapitation and teratozoospermia 2. The gene has been identified as a candidate for intellectual disability in some populations 3 and shows altered methylation patterns in chr17 mosaicism 4. These findings establish CNTROB as crucial for centrosome function, ciliogenesis, and male fertility.