Figure 2.

Hyls1 D226G leads to centriole integrity defects. (A–L) Left: Representative images of centriole proximal end (CEP135), centrioles (Ac-TUBULIN), and distal appendages (CEP164) analyzed by U-ExM in kidneys (A), trachea (C), lungs (E), heart (G), brain (I), and thymus (K) in control (Hyls1+/+ or Hyls1+/DG) and Hyls1DG/DG P0 animals. Right: Quantification of centriole defects and centriole asymmetry in kidneys (B), trachea (D), lungs (F), heart (H), brain (J), and thymus (L) in control (Hyls1+/+ or Hyls1+/DG) and Hyls1DG/DG P0 animals. N = 3 mice per genotype. Data are represented as mean ± SEM. Statistical significance was assessed using an unpaired two-tailed Student’s t test with Welch’s correction for centriole asymmetry analysis. (*) P < 0.05, (***) P < 0.001. Only significant results are indicated. Centriole defects analysis was assessed using two-way ANOVA with post-hoc analysis and results are summarized in supplementary material. Scale bar is 250 nm. Asterisk (*) indicates defective centrioles.

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