FIGURE

Fig. 1

ID

ZDB-FIG-170424-42

Publication

Smith et al., 2017 - A novel early onset phenotype in a zebrafish model of merosin deficient congenital muscular dystrophy

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Fig. 1

Caf mutants display a coiling phenotype as early as 23 hpf.

A) One full coil completed by a 24 hpf wild type (lama2^+/+ and lama2^+/-; WT) zebrafish embryo. B) Number of coils of WT siblings and caf (lama2^-/-) mutants over 30 seconds when left in their chorions. There is no significant difference in the coiling abilities of caf mutants and their WT siblings at 22 hpf (WT sibs: 1.071 ± 0.3391, n = 14; cafs: 1.000 ± 0.7071, n = 4; p>0.9999), 23 hpf (WT sibs: 0.9286 ±0.3987, n = 14; cafs: 1.000 ± 1.000, n = 2; p>0.9999) or 24 hpf (WT sibs: 1.133 ± 0.2906, n = 15; cafs: 1.222 ± 0.3643, n = 9; p>0.9999). C) Number of coils of WT siblings and caf mutants in the 15 seconds immediately after manual dechorionation. At 22 hpf, there was no significant difference in number of coils completed by WT siblings and caf mutants (WT sibs: 3.654 ± 0.7685, n = 26; cafs: 3.250 ± 1.031, n = 8; p>0.9999). However, cafs completed significantly less coils than their WT sibs at 23 hpf (WT sibs: 7.444 ± 0.5587, n = 72; cafs: 3.476 ± 0.5921, n = 21; p = 0.0194) and 24 hpf (WT sibs: 10.71 ± 0.4436, n = 163; cafs: 2.936 ± 0.6646, n = 47; p<0.0001). Bars represent mean ± SEM.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	lama2^{teg15a/teg15a}
Observed In:	musculoskeletal movement thigmotaxis
Stage Range:	26+ somites to Prim-5

Phenotype Detail

Acknowledgments

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