HYLS1 interacts with CEP120. (A) Co-immunoprecipitation of FLAG-CPAP with HYLS1^WT-mCherry or HYLS1^DG-mCherry in HYLS1^−/− DLD1 cells. Cells were induced with doxycycline for 4 days and FLAG-CPAP transfected on the second day for 48 h. (B) Co-immunoprecipitation of 6xHA-TUBB5 with HYLS1^WT-mCherry or HYLS1^DG-mCherry in HYLS1^−/− DLD1 cells. Cells were induced with doxycycline for 4 days and 6xHA-TUBB5 transfected on the second day for 48 h. (C) Co-immunoprecipitation of 6xHA-CEP120 with HYLS1^WT-mCherry or HYLS1^DG-mCherry in HYLS1^−/− DLD1 cells. Cells were induced with doxycycline for 4 days and 6xHA-CEP120 transfected on the second day for 48 h. (D) A model for how impaired recruitment of HYLS1 D211G to the centriole causes the cilia defects underlying hydrolethalus syndrome. Source data are available for this figure: SourceData F7.