FIGURE

Fig. 2

ID

ZDB-FIG-180312-3

Publication

Li et al., 2017 - Rapid functional analysis of computationally complex rare human IRF6 gene variants using a novel zebrafish model

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Fig. 2

Periderm rupture and craniofacial development can be rescued by injection of either zebrafish or human wild type IRF6 mRNA.

(A-L) Zebrafish embryos at 96 hpf stained with alcian blue for cartilaginous craniofacial elements. Maternal/zygotic-null irf6 ^-/- embryos were rescued by microinjection of either zebrafish irf6 mRNA (E-H) or human IRF6 mRNA (I-L) at the one-cell stage, preventing the periderm rupture phenotype and restoring normal craniofacial development compared to wild type embryos (A-D). Scale bar = 150 μm. (M-N) Dimensional measurements of dissected ethmoid plates at 96 hpf, with length (l) and width (w) denoted by dashed lines on panel (D). The length (M) and width (N) of ethmoid plates from zebrafish and human IRF6 mRNA rescued maternal/zygotic-null irf6 ^-/- embryos are statistically indistinguishable in dimensions compared those of wild type embryos. Error bar = 2xSEM, n = 12.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Phenotype Detail

Acknowledgments

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