Given the effect on ciliary length control in adult-onset ADPKD in an ANKMY2-dependent manner (see next section), and ciliary length control being mediated by factors that localize in the cilia-centrosomal complex [34,35], the most parsimonious model would be that the effect on ciliary/peri-ciliary adenylyl cyclase signaling by ANKMY2 is the major contributing factor in regulating cystic burden in ADPKD (Fig 8). The gene discussed is ANKMY2; the disease is autosomal dominant polycystic kidney disease.