FIGURE

Fig 4

ID

ZDB-FIG-210821-37

Publication

Kuil et al., 2021 - Size matters: Large copy number losses in Hirschsprung disease patients reveal genes involved in enteric nervous system development

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Fig 4

Validation of tbx2, ufd1l and mapk8 gene disruption shows effects on the ENS in zebrafish.

(A) Figures and graphs showing that disruption of tbx2a/b in zebrafish does not result in overall significantly reduced numbers of phox2bb-GFP+ cells, however it does induce ENS phenotypes in category II and III in a significant number of zebrafish larvae. (B) Figures and graphs showing that disruption of ufd1l in zebrafish results in significantly reduced numbers of phox2bb-GFP+ cells and increase in the percentage of fish presenting with an ENS phenotype. (C) Figures and graphs showing that disruption of mapk8a/b in zebrafish does not result in overall significantly reduced numbers of phox2bb-GFP+ cells, however it does induce ENS phenotypes in a significant number of zebrafish larvae. Statistical tests used: students t-test and "N-1" Chi-squared test; * p < 0.05 ** p < 0.01 *** p < 0.001; Exact p-values and statistics can be found in S11 Table.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	w37Tg + CRISPR1-ufd1l w37Tg + CRISPR1-tbx2a + CRISPR1-tbx2b w37Tg + CRISPR2-mapk8a + CRISPR2-mapk8b
Knockdown Reagents:	CRISPR1-tbx2a CRISPR1-tbx2b CRISPR1-ufd1l CRISPR2-mapk8a CRISPR2-mapk8b
Observed In:	enteric nervous system development enteric neuron posterior intestine
Stage:	Day 5

Phenotype Detail

Acknowledgments

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