FIGURE
Fig. 4
- ID
- ZDB-FIG-220905-10
- Publication
- Lin et al., 2022 - Biallelic variants in WARS1 cause a highly variable neurodevelopmental syndrome and implicate a critical exon for normal auditory function
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Fig. 4
Zebrafish wars1 F0 mutants displayed craniofacial dysmorphism and skeletal muscle myopathy. (a) Alcian blue staining was performed to reveal the craniofacial cartilage structure of uninjected control, wars1 F0, and rescue larvae at 8 dpf. Ethmoid plate (ep), Meckel's cartilage (Mk), palatoquadrate (pq), and ceratohyal (ch). Scale bar = 200 μm. (b) Quantification of the length of Meckel's cartilage measurement as indicated in control, F0 mutants, and mutants with WARS1 mRNA rescue. (c) Quantification of alignment of palatoquadrate and ceratohyal cartilage in controls, F0 mutants, and mutants with WARS1 mRNA rescue. (d) Analysis of muscle development using phalloidin stain that labels actin filaments. 8 dpf old uninjected control embryos showing well-developed actin fibers (left panel), wars1 F0 mutants showing disorganized actin fibers (middle panel), and wars1 mutant phenotypes rescued the phenotype with human WARS1 mRNA (right panel). Scale bar = 50 μm. mRNA, messenger RNA. For (b, c), each group contains 10 larvae and each dot represents one larvae. Error bar = mean ± SD. One-way ANOVA with Tukey's multiple comparisons test: ****p < 0.0001. All marked groups were compared to F0.
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Expression Data
Expression Detail
Antibody Labeling
Phenotype Data
| Fish: | |
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| Knockdown Reagents: | |
| Observed In: | |
| Stage: | Days 7-13 |
Phenotype Detail
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Full text @ Hum. Mutat.