FIGURE

Fig. 5

ID

ZDB-FIG-211119-7

Publication

Ziegler et al., 2021 - Bi-allelic variants in IPO8 cause a connective tissue disorder associated with cardiovascular defects, skeletal abnormalities, and immune dysregulation

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Fig. 5

pSMAD nuclear translocation defects in ipo8−/− embryos highlight impaired TGF-β/BMP signaling (A) Maximal projections of confocal Z stacks of WT and ipo8−/− embryos labeled for pSmad1/5/9 (green) and DAPI (blue) showing well-defined ventral to dorsal gradient of pSmad5 in WT gastrulating zebrafish embryos (left panel) but not in the ipo8−/− embryos (right panel). Scale bar represents 100 μm. (B) Confocal images of the ventral sides of WT and ipo8−/− embryos labeled for pSmad1/5/9 (green), phalloidin (red), and DAPI (blue). Arrowheads highlight the membrane localization of the pSmad staining in the ipo8−/− mutants. Scale bar represents 20 μm. Dot plots representative of three experiments show quantifications of the ratio of pSmad1/5/9 cytoplasmic signal over nuclear signal; 5 WT and 6 ipo8−/− embryos were analyzed. Median and IQR are shown. p values were calculated by Mann Whitney test (∗∗p < 0.01). (C) Heatmaps of differentially expressed genes between WT and ipo8−/− embryos at 13 and 24 hpf. Three to four biological replicates are shown per group. The top enriched gene ontology term for biological process is highlighted for each cluster.

Expression Data

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Antibody Labeling

Phenotype Data

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Acknowledgments

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