ZFIN Image: Otten et al., 2018, Fig. 1

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Figure Caption

Fig. 1

Small‐molecule drug screens identify compounds relevant for CCM

A. Overview of the four different screening assays used in this study. Zebrafish embryos and C. elegans are screened in 24‐well and 96‐well plates, respectively. The most promising active compounds are retested in shCCM2 HUVECs. One compound is tested for suppression of vascular lesion formation in the cerebellum of iCCM2 and iCCM3 mouse models.
B. Overlap of rescue compounds screened in the different assays.
C–E Examples of rescue of cardiovascular defects of the zebrafish ccm2^m201 mutant. Inverted images of confocal z‐scan projections of the 46 hpf head region and heart (endocardium) of wild‐type (WT) and ccm2^m201 mutant zebrafish embryos carrying the endothelial Tg(kdrl:GFP)^s843 reporter transgene. Embryos are untreated (C) or treated between 17 and 48 hpf with 10 μM of the Lck inhibitor C8863 (D) or with 10 μM of the ERK5 inhibitor XMD8‐92 (E). Both compounds resulted in a reduction in heart size and narrowing of the heart tube at the atrioventricular canal (arrowheads). Scale bar is 100 μm.

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Phenotype details

Acknowledgments

This image is the copyrighted work of the attributed author or publisher, and ZFIN has permission only to display this image to its users. Additional permissions should be obtained from the applicable author or publisher of the image. Full text @ EMBO Mol. Med.