Centrosomal protein of 131kDa (CEP131) is a cilium-specific protein required for cilium/flagellum formation []. In Drosophila, CEP131, also known as Azi1, functions in ciliogenesis and is involved in genome stability and centriole duplication [, ]. In mice, it mediates trafficking events necessary for spermiogenesis and male fertility []. It interacts with BBS4 (Bardet-Biedl syndrome 4) and regulates ciliary trafficking of the BBSome [].
