In order to decipher the impact of CEP120 ciliopathy mutations on centriole distal ends, we assayed the localization of CEP164, a distal appendage protein present on mother centrioles (Graser et al., 2007a), and that of TALPID3, a protein enriched at the extreme distal ends of mother and daughter centrioles (Kobayashi et al., 2014, Yin et al., 2009). Here, CEP164 is linked to ciliopathy.