FIGURE

Figure 4

ID

ZDB-FIG-231003-12

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 4

Loss of Flii results in a reduced number of cardiac trabeculae.

(A) Single confocal planes of representative Tg(myl7:LIFEACT-GFP) ventricular outer curvatures at 96 hpf through 6 dpf of flii^+/? siblings (left) and flii^{D110fs/D110fs} mutants (right). The 96 hpf hearts express Tg(myl7:nDsRed2) to visualize nuclei. Yellow false color depicts outer curvature. Trabecular myocardium is highlighted in blue. Scale bars: 25 μm. (B) Cropped areas corresponding to white rectangles in A. The dotted lines depict the compact layer of cardiomyocytes. (C) Quantification of protruding cardiomyocytes out of the single-layered compact myocardium in the ventricular outer curvature from sagittal planes at 96 hpf through 6 dpf. Unpaired t test; values represent means ± SEM; (96 hpf flii^+/? siblings n = 6, flii^{D110fs/D110fs}n = 9); (120 hpf flii^+/? siblings n = 5, flii^{D110fs/D110fs}n = 5); (6 dpf flii^+/? siblings n = 3, flii^{D110fs/D110fs}n = 3).

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Phenotype Detail

Acknowledgments

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