FIGURE

Fig 4

ID

ZDB-FIG-201107-4

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 4

Knockdown of Bag3 leads to heart and skeletal muscle dysfunctions only in bag^+/+and bag3^+/-embryos. (A) Brightfield and birefringence images of bag3^+/+, bag3^+/- and bag3^-/- embryos at 72 hpf injected with MO-bag3. Densitometric analysis of birefringence (n = 4). Representative samples are shown (One-way ANOVA followed by tukey's multiple comparison analysis, P = 0.0012). (B) Only bag3^-/- embryos injected with MO-bag3 do not show (cardio-)myopathy, whereas bag3^+/+and bag3^+/- develop the characteristic bag3 morphant phenotype (N = 3, n = 40, mean ± S.D., P<0.0001, two-tailed value for Fisher´s exact test. bag3^+/+ and bag3^+/- P = 0.5346; bag3^+/+ and bag3^-/- P<0.0001; bag3^+/- and bag3^-/- P<0.0001). (C) Tropomyosin immunostainings of bag3^+/+, bag3^+/-, and bag3^-/- embryos injected with MO-bag3 at 72 hpf reveal that only bag3^-/- mutant embryos injected with MO-bag3 does not develop muscle fiber disruptions. (D) Heart rate quantification at 72 hpf reveals impairments only in bag3^+/+and bag3^+/- embryos injected with MO-bag3 (N = 3, n = 9, mean ± S.D. One-way ANOVA followed by tukey's multiple comparison analysis, P = 0.0008). (E) Quantification of ventricular FS at 72 hpf reveals contractile dysfunctions only in bag3^+/+and bag3^+/- embryos injected with MO-bag3 (N = 3, n = 9, mean ± S.D. One-way ANOVA followed by tukey's multiple comparison analysis, P<0.0001). (F)bag3^-/- embryos injected with MO-bag3 show proper flight response upon mechanical stimulus, whereas bag3^+/+and bag3^+/- embryos injected with MO-bag3 do not show an adequate touch response (N = 3, n = 60, mean ± S.D. One-way ANOVA followed by tukey's multiple comparison analysis, P<0.0001).

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	bag3^{ulm104/ulm104} + MO2-bag3 WT + MO2-bag3 bag3^ulm104/+ + MO2-bag3
Knockdown Reagent:	MO2-bag3
Observed In:	heart contraction response to mechanical stimulus skeletal muscle whole organism muscle
Stage:	Protruding-mouth

Phenotype Detail

Acknowledgments

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