Hyls1 D226G leads to smaller animals with kidney developmental defects. (A) Schematic representation of the strategy used to generate the Hyls1 D226G mouse model. (B–D) Embryo size and weight at E14.5 (B), E18.5 (C), and P0 (D) developmental stages in Hyls1^+/+, Hyls1^+/DG, and Hyls1^DG/DG animals. N ≥ 4 mice per genotype. (E) Cerebral cortex (left) and midbrain (right) area analysis in Hyls1^+/+, Hyls1^+/DG, and Hyls1^DG/DG P0 animals. Dotted circles indicate the data point used for the representative images in F. N ≥ 7 mice per genotype. (F) Representative images from the brain size analysis in E. (G) Histological analysis of kidneys in Hyls1^+/+, Hyls1^+/DG, and Hyls1^DG/DG P0 animals. Asterisks (*) represent fibrosis and enlarged tubules in Hyls1^DG/DG animals. Data are represented as mean ± SEM. Statistical significance was assessed using one-way ANOVA with Tukey’s multiple comparisons test (B–E). (*) P < 0.05, (**) P < 0.01, (****) P < 0.0001. Only significant results are indicated.