How CEP164 ciliopathy mutations impair ciliogenesis

CEP164 recruits TTBK2 to the distal end of centrioles to allow primary cilium formation. In this issue of Structure, Rosa e Silva et al. (2022) present co-crystallized structures that show the molecular basis of this recruitment and define how ciliopathy mutations in CEP164 disrupt the CEP164-TTBK2 complex.

Automated summary

In this study, the co-crystallized structures revealed the molecular mechanism of CEP164 recruiting TTBK2 to the distal end of centrioles and how ciliopathy mutations in CEP164 disrupt the CEP164-TTBK2 complex.