FIGURE

Figure 7

ID

ZDB-FIG-231003-15

Publication

Ruijmbeek et al., 2023 - Bi-allelic variants in FLII cause pediatric cardiomyopathy by disrupting cardiomyocyte cell adhesion and myofibril organization

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Figure 7

Aberrant activation of the Notch and Hippo signaling pathways in Flii-deficient ventricles.

(A) Representative 3D volume renderings of TP1bglob:VenusPEST of flii^+/? sibling and flii^{D110fs/D110fs} hearts at 96 hpf. White dotted line outlines the heart. Note that there is no Notch reporter expression in the ventricle of flii^{D110fs/D110fs} hearts, whereas there is expression in their AVC and OFT. Each group n = 5. V, ventricle; A, atrium; AVC, atrioventricular canal; OFT, outflow tract. (B) Representative maximum-intensity projections of wholemount flii^+/? sibling and flii^{D110fs/D110fs} hearts at 60 hpf stained for Wwtr1. flii^+/? siblings n = 7, flii^{D110fs/D110fs}n = 11. (C) Corresponding confocal sagittal sections of the wholemount ventricles shown in B. Nuclei are counterstained with DAPI, and cardiomyocyte F-actin myofibrils are marked with myl7:LIFEACT-GFP expression; scale bars, 20 μm.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Phenotype Detail

Acknowledgments

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