FIGURE

Fig. 3

ID

ZDB-FIG-180828-63

Publication

Tsai et al., 2018 - Small molecule inhibition of RAS/MAPK signaling ameliorates developmental pathologies of Kabuki Syndrome

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Fig. 3

Compound 8 ameliorates the CE and jaw defects caused by loss of rap1 and kdm6a. (a) gRNA targeting both zebrafish rap1a and rap1b were injected into embryos at the 1-cell stage with Cas9 protein. Larvae were incubated with compound 8 (100 nM) at the 8-cell stage and scored for micrognathia. 5 dpf larvae were stained with Alcian blue to assess the layout of the jaw cartilage. In comparison to control embryos (top), rap1-CRISPR embryos have smaller jaws. This phenotype can be ameliorated by treatment with compound 2 and 8. (b) Quantitative measurement of the distance between MK and CH cartilages. (c) Larvae were scored for CE (n > 30 for each concentration). (d) 5 dpf embryos were stained with Alcian blue to assess the layout of jaw cartilage. In comparison to control embryos (top), kdm6a-MO embryos exhibit smaller jaws. This phenotype can be ameliorated by treatment with compound 8. (e) Quantitative measurement of the distance between MK and CH cartilages. *p < 0.05; ***p < 0.001. Error bars show SEM (standard error of the mean).

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	EKW + MO2-kdm6a EKW + CRISPR1-rap1ab + CRISPR1-rap1b
Condition:	chemical treatment by environment: B-Raf inhibitor
Knockdown Reagents:	CRISPR1-rap1ab CRISPR1-rap1b MO2-kdm6a
Observed In:	ceratohyal cartilage convergent extension involved in gastrulation mandibular arch skeleton Meckel's cartilage
Stage Range:	Prim-5 to Day 5

Phenotype Detail

Acknowledgments

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