FIGURE

Figure 3

ID

ZDB-FIG-250519-3

Publication

Munro et al., 2025 - Deoxynucleoside supplementation ameliorates the disease associated phenotypes in a zebrafish model of RRM2B mtDNA depletion syndrome

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Figure 3

Deoxynucleoside supplementation in 5 dpf rrm2b and wild type larvae. A. Relative mtDNA copy number in 5 dpf rrm2b larvae treated between 0 dpf and 5 dpf with 50 μM of deoxynucleosides: dAdo (A), dGuo (G), dCtd (C) & dTmd (T) with 5 μM adenosine deaminase inhibitor, EHNA. Untreated 5 dpf rrm2b^+/+ & rrm2b^+/− tail muscle (n = 54) (circles with thin border), treated 5 dpf rrm2b^+/+ & rrm2b^+/− tail muscle (n = 36) (circles with thick border). Untreated 5 dpf rrm2b^−/− tail muscle (n = 21) (squares with thin border), treated 5 dpf rrm2b^−/− tail muscle (n = 20) (squares with thick border). Results are shown as mean ± SEM (statistical significance was determined with a Mann–Whitney U test, ^*^*^*^*P < 0.0001). B–E. Relative mtDNA copy number of pools of 10, 5 dpf WT whole larvae treated with 15 combinations of deoxynucleosides (circles with thick border) or untreated (circles with thin border). 5 μM of ENHA (deaminase inhibitor) was also supplemented with each deoxynucleoside combination. 3 pools of 10 larvae per treatment condition. Results are shown as mean ± SEM (statistical significance was determined with a one-way ANOVA, ^* = adj. P < 0.05).

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Phenotype Detail

Acknowledgments

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