FIGURE

Fig. 10

ID

ZDB-FIG-230317-13

Publication

Smith et al., 2021 - Lysosomes and the pathogenesis of merosin-deficient congenital muscular dystrophy

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Fig. 10

UPS is upregulated and MG-132 treatment improves the swimming behavior and dystrophic phenotype of caf mutants. (A and B) Western blot analysis showing increased levels of Polyubiquitinylated proteins (PolyU) in caf mutants. (C–E) Swimming behavior analysis. (C) There is no significant improvement in time spent swimming in caf mutants treated with 10 μM MG132 (n = 166). (D and E) There are significant improvements in distance traveled and average velocity in caf mutants at 3 dpf after treatment with 10 μM MG132. (F) Treatment with 10 μM MG132 improves the dystrophic phenotype of caf mutants at 3 dpf but does not restore muscle integrity to WT levels (n = 43). ^**P < 0.0075; ^****P < 0.0001. Bars represent mean ± SEM.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	lama2^{teg15a/teg15a}
Condition:	chemical treatment: N-benzyloxycarbonyl-L-leucyl-L-leucyl-L-leucinal
Observed In:	muscle protein ubiquitination swimming behavior
Stage Range:	Protruding-mouth to Day 5

Phenotype Detail

Acknowledgments

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