Figure 1

Figure 1

Examples of experimental approaches used for the phenotypical analysis of the zebrafish LAMA2-related congenital muscular dystrophy (CMD, LAMA2-MD) model. (A,A’) Birefringence assay. The organization of muscle fibers can be seen by polarized light. Detached muscle fibers in the caf mutant show up as dark regions in the muscle (arrows). (B,B’) Whole-mount staining. Phalloidin stains the actin filaments in the muscle. Muscle fibers detached from the myotendinous junctions (MTJs) in the caf mutant can be easily identified (arrows). (C,C’) Injected fluorescent marker. unc53:mCherry-CAAX-pA construct (Zhao et al., 2019), which marks muscle cells, was injected into 1-cell stage embryos and visualized by live imaging at 3 dpf. Detached fibers in the caf mutant can be easily identified (arrows). (D,D’) Swimming assay. Swim behavior can be tracked and quantified using Viewpoint Zebrabox software (Viewpoint Life Sciences Inc.). Time spent moving, distance traveled and speed of movement are useful indicators of muscle function. Fewer tracks and lower speed (indicated by green tracks) are seen in the recorded caf mutants.