FIGURE

Fig. 1

ID
ZDB-FIG-200129-11
Publication
Sun et al., 2020 - Dstyk mutation leads to congenital scoliosis-like vertebral malformations in zebrafish via dysregulated mTORC1/TFEB pathway
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Fig. 1

<italic>Smt</italic> mutation leads to CS-like vertebral malformations.

a Bright-field images showing shortened somites and body length in smt mutant at 40 hpf. The bottom panel showed enlarged parts. Red line marks single somites. b Graph depicting the body length measurements of WT (blue line) and smt mutant (red line) from 1.5 dpf to 6.5 dpf, n = 20 independent embryos for WT and smt mutant. Data points represent average body length and error bars represent standard deviation. c Lateral (top) and dorsal views (bottom) showed slight wavy bending in smt mutant at 7 dpf. d At about 20 dpf, smt mutant showed different degrees of curve severity in dorsal view: mild (top), moderate (middle) and severe (bottom) curvature. e Whole mount image of 3-month-old smt mutant (bottom) and WT (top). Smt mutants had scoliosis and quantification of the body length were much shorter than WT (f), n = 5 independent zebrafishes for WT and smt mutant. ** p < 0.01. p values were determined by unpaired two-tailed Student’s t-test. X-rays (g) and three-dimensional micro-CT (h) revealed severe scoliosis and kyphosis in smt mutant (bottom). Boxed regions are magnified in the right panel in (h). Data are presented as mean ± SD. Scale bar represent 200 μm in (a), 400 μm in (c), 1 mm in (d and h), 2 mm in (d). Source data are provided as a Source Data file.

Expression Data

Expression Detail
Antibody Labeling
Phenotype Data
Fish:
Observed In:
Stage Range: Prim-25 to Adult

Phenotype Detail
Acknowledgments
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