FIGURE

Fig. 7

ID

ZDB-FIG-230317-10

Publication

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

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Fig. 7

Treatment with the lysosomal agonist MLSA1 partially improves swimming behavior of caf mutants at 5 dpf. (A) MLSA1 reduces swimming behavior of WT zebrafish at 5dpf in a dose-dependent manner. Conversely, in caf mutants, treatment with 5 or 8 μM significantly increases the time spent moving. However, there was no significant improvement in the total distance traveled or average velocity in treated versus untreated caf mutants at any dose tested (n = min 56). Bars represent mean ± SD. (B) Confocal micrographs of whole-mount embryos stained with anti-Lamp1 antibody (green), Phalloidin (magenta) and DAPI (blue). There are no differences in Lamp1 distribution between treated and untreated groups at 5 dpf, and no evidence of increased fiber integrity or decreased detachment.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Phenotype Detail

Acknowledgments

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