Gross phenotypes of Zswim8−/− mice. (A) Organization of the murine Zswim8 genomic locus. Shown below the gene model (thick gray boxes indicate the exonic coding sequence; thin gray boxes, exonic untranslated regions; gray lines, introns; blue box, Cas9 PAM; yellow box, annotated BC box motif; pink box, annotated Cullin box motif) are the genomic DNA sequences of the wild-type (WT) allele and four mutant alleles (“–1,” “–7,” “–4,” and “+2”), as well as the amino acid sequence of the expected WT translation product. Blue text indicates the WT protein sequence; black text, WT DNA sequence; red text, insertions; and red dashes, deletions. (B) Penetrant lethality of Zswim8−/− animals. Shown are genotypes of offspring produced from intercrosses between Zswim8+/− parents. Offspring were counted at either embryonic day (E)18.5, postnatal day (P) 0, or P21. Differences from the Mendelian expectation were evaluated using the chi-square test (two-tailed): (n.s.) not significant, P > 0.05; (****) P < 0.0001. (C) Cyanotic phenotype of Zswim8−/− pups. Shown are P0 neonate siblings with the indicated genotypes, cropped from the same photograph. (D) Smaller size of Zswim8−/− embryos. Plotted are weights of E18.5 embryos produced from intercrosses between Zswim8+/− parents. Horizontal lines indicate the mean. Significance of differences were evaluated using ANOVA (Tukey's multiple comparisons test): (n.s.) not significant, P > 0.05; (****) P < 0.0001.
