The majority of ciliopathic mutations target components of the cilium, but mutations in a number of centrosomal genes, such as CEP164, TALPID3, CSPP1, PLK4, and CEP120, also cause ciliopathies, raising the question of how their dysfunction mechanistically translates to the corresponding disease (Alby et al., 2015, Chaki et al., 2012, Martin et al., 2014, Roosing et al., 2016, Shaheen et al., 2014, Shaheen et al., 2015, Tuz et al., 2014). This evidence concerns the gene CEP164 and ciliopathy.