Hyls1 D226G does not impair MCC differentiation. (A) Schematic representation of approach used to score the centriole microtubule wall asymmetry (AS). LMT: longest microtubule; SMT: shortest microtubule. (B) Schematic representation of the calculations used to score centriole defects (long, normal, broken, and short). (C) Quantification of longest microtubule length (LMT) and shortest microtubule length (SMT) in the kidneys, trachea, lungs, heart, brain, and thymus of control (Hyls1+/+ or Hyls1+/DG) and Hyls1DG/DG P0 animals by U-ExM. N = 3 mice per genotype. (D) U-ExM of MCCs in the ependyma of the lateral ventricles in the brain of control (Hyls1+/+ or Hyls1+/DG) and Hyls1DG/DG P0 animals. (E) U-ExM analysis of MCCs in the choroid plexus in the brain of control (Hyls1+/+ or Hyls1+/DG) and Hyls1DG/DG P0 animals. (F) Immunofluorescence analysis of MCCs in the choroid plexus in the brain of (Hyls1+/+ or Hyls1+/DG) and Hyls1DG/DG P0 animals. Data are represented as mean ± SEM. Statistical significance was assessed using one-way ANOVA with Tukey’s multiple comparisons test (C). (*) P < 0.05, (**) P < 0.01. Only significant results are indicated. Scale bar is 1 μm in D and E and 10 μm in F.