FIGURE

Figure 3

ID
ZDB-FIG-231204-36
Publication
Serrano et al., 2023 - Genetic model of UBA5 deficiency highlights the involvement of both peripheral and central nervous systems and identifies widespread mitochondrial abnormalities
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Figure 3

Survival, motor function and growth are impaired in uba5ex1s/ex1s fish. (A) 25 uba5+/+, 25 uba5+/ex1s and 25 uba5ex1s/ex1s embryos were raised, and their survival was analysed. (B) Quantification of the distance travelled showed a significant reduction of the motor function in uba5ex1s/ex1s compared to wild-type siblings. Data are represented as mean ± SD predicted by the linear model statistical test for one biological replicate analysed at 14 (n = 23 uba5+/+; n = 25 uba5+/ex1s; n = 23 uba5ex1s/ex1s), 21 (n = 23 uba5+/+; n = 25 uba5+/ex1s; n = 14 uba5ex1s/ex1s), 28 (n = 23 uba5+/+; n = 23 uba5+/ex1s; n = 12 uba5ex1s/ex1s) and 35 dpf (n = 23 uba5+/+; n = 23 uba5+/ex1s; n = 6 uba5ex1s/ex1s). (C) Measurement of the body length at 5 dpf did not reveal a significant difference between uba5ex1s/ex1s and wild-type siblings (n = 9 uba5+/+; n = 13 uba5ex1s/+; n = 6 uba5ex1s/ex1s). Each dot represents an individual zebrafish. The error bars represent mean ± SD for one biological replicate. n.s., non-significant using a one-way ANOVA. (D) Measurement of the body length at 14 (n = 23 uba5+/+; n = 25 uba5+/ex1s; n = 23 uba5ex1s/ex1s), 21 (n = 21* uba5+/+; n = 25 uba5+/ex1x; n = 14 uba5ex1s/ex1s), 28 (n = 23 uba5+/+; n = 21* uba5+/ex1s; n = 12 uba5ex1s/ex1s) and 35 dpf (n = 23 uba5+/+; n = 23 uba5+/ex1s; n = 5* uba5ex1s/ex1s) demonstrates that uba5ex1s/ex1s are smaller than wild-type siblings at later stages of development. Data are represented as mean ± SD predicted by the linear model statistical test. *These numbers were originally the same as in (B), but we were unable to measure the body length of some fish, and therefore, these were not included in the analysis.

Expression Data

Expression Detail
Antibody Labeling
Phenotype Data
Fish:
Observed In:
Stage Range: Day 5 to Days 45-89

Phenotype Detail
Acknowledgments
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