FIGURE

Fig 2

ID

ZDB-FIG-201107-2

Publication

Diofano et al., 2020 - Genetic compensation prevents myopathy and heart failure in an in vivo model of Bag3 deficiency

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Fig 2

Genetic loss of <italic>bag3</italic> does not interfere with heart and skeletal muscle function.

(A) Lateral view of brightfield and birefringence images for bag3^+/+, bag3^+/- and bag3^-/- embryos at 72 hpf exhibiting homogenous birefringence signals. (B) Densitometric analysis of birefringence does not report muscle disorganization in bag3^-/- embryos. Representative samples are shown (n = 6, One-way ANOVA followed by tukey's multiple comparison analysis, P = 0.9688). (C) Embryos at 72 hpf reveal no myopathic phenotype (N = 3, n = 100, two-tailed value for Fisher´s exact test. bag3^+/+ and bag3^+/- P = 0.8096; bag3^+/+ and bag3^-/- P = 0.4949; bag3^+/- and bag3^-/- P = 0.1925). (D) Heart rate quantification at 72 hpf reveals no impairments under physiological conditions (N = 3, n = 12, mean ± S.D. One-way ANOVA followed by tukey's multiple comparison analysis, P = 0.6026). (E) Quantification of ventricular FS at 72 hpf does not reveal contractile dysfunction (N = 3, n = 12, mean ± S.D. One-way ANOVA followed by tukey's multiple comparison analysis, P = 0.5061). (F) Quantification of the touch evoked assay. bag3^-/- embryos reveal no significant difference in responsiveness upon mechanical stimulus (N = 3, n = 90, mean ± S.D. One-way ANOVA followed by tukey's multiple comparison analysis, P = 0.7851). (G) Immunostaining of bag3^+/+, bag3^+/- and bag3^-/- embryos at 72 hpf, with sarcomeric Tropomyosin, showing no muscle fiber disruptions. (H) Brightfield and birefringence images of mutant embryos at 120 hpf under stressed conditions reveal no myopathic phenotype, which was confirmed by densitometric analysis of birefringence signals. Representative samples are shown (n = 4, One-way ANOVA followed by tukey's multiple comparison analysis, P = 0.1824). (I) Embryos, under stressed conditions, do not develop myopathic phenotype (N = 4, n = 50, two-tailed value for Fisher´s exact test. bag3^+/+ and bag3^+/- P = 0.6050; bag3^+/+ and bag3^-/- P = 0.8320; bag3^+/- and bag3^-/- P = 0.8582). (J) Immunostaining of bag3^+/+, bag3^+/- and bag3^-/- embryos at 120 hpf under stressed conditions (2 hours in 1% methylcellulose), with sarcomeric Tropomyosin, showing no muscle fiber disruptions.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	WT bag3^{ulm104/ulm104} bag3^ulm104/+
Condition:	chemical treatment by environment: methyl cellulose, mechanical stress
Observed In:	heart contraction response to mechanical stimulus whole organism muscle
Stage Range:	Protruding-mouth to Day 5

Phenotype Detail

Acknowledgments

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