ZFIN Image: Solman et al., 2022, Figure 1

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Figure Caption

Figure 1

Shp2^D61G zebrafish display Noonan syndrome (NS)-like traits.

(A) Sequencing trace derived from Shp2^D61G/D61G zebrafish. Oligonucleotide sequence used to generate the model is underlined. Nucleotide substitutions for D61G mutation (red), silent mutations close to the PAM site (green), and the PAM site (blue) are indicated. (B) Immunoblot of Shp2 levels from five pooled Shp2a^wtShp2b^-/- or Shp2a^D61G/D61GShp2b^-/- embryos using antibodies for Shp2 and tubulin (loading control) (Figure 1—source data 1). (C) Representative images of typical Shp2^D61G zebrafish embryonic phenotypes at 5 days post fertilization (dpf). Blue arrows: jaw, green arrows: heart, red arrows: swim bladder. (D) Quantification of phenotypes of Shp2^wt, Shp2^D61G/wt, and Shp2^D61G/D61G embryos scored as in (C) normal, mild, and severe. (E) Body axis length of Shp2^wt, Shp2^D61G/wt, and Shp2^D61G/D61G embryos at 5 dpf. (F) Representative images of Alcian blue-stained head-cartilage of 4dpf Shp2^wt and Shp2^D61G/wt embryos (x) width of ceratohyal, (y) distance to Meckel’s cartilage. (G) Quantified craniofacial defects (x/y ratio). (H) Representative ventricular kymographs derived from high-speed video recordings of beating hearts of 5 dpf Shp2^wt and Shp2^D61G/wt embryos. Red dotted lines indicate one heart period. (I) Heart rates derived from the ventricular kymographs. (J) Representative images of typical Shp2^D61G zebrafish adult phenotypes at 24 weeks post fertilization (wpf). Red arrows indicate skin redness in the jaw region. Scale bar, 0.5 cm. Insets, zoom-in of boxed regions. (K) Body axis lengths of 10 Shp2^wt, 25 Shp2^D61G/wt, and 10 Shp2^D61G/D61G zebrafish measured weekly between 5 dpf and 20 wpf of age. (D,E,G,I) Measurements originate from three distinct experiments. Number on bars: number of embryos. (E,G,I,K) Error bars: standard error of the mean (SEM), *p < 0.05; **p < 0.01, ***p < 0.001, ANOVA complemented by Tukey’s HSD.

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Figure 1

Figure 1

Shp2D61G zebrafish display Noonan syndrome (NS)-like traits.

Shp2^D61G zebrafish display Noonan syndrome (NS)-like traits.