While Kif3a (Lin et al, 2003) and Cep164 (Airik et al, 2019) deletion can cause cystogenesis due to a lack of primary cilia in neonatal contexts, in adult models of autosomal dominant PKD, Ma et al have demonstrated a cilia-dependent cyst activation pathway (CDCA) where a structurally intact cilia transmits an aberrant growth signal which cilia removal via Kif3a co-deletion prevents (Ma et al, 2017). Here, CEP164 is linked to cyst.