FIGURE

Figure 5

ID

ZDB-FIG-250519-5

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 5

Assessment of purine deoxynucleoside supplementation in 7 dpf rrm2b larvae—. (A) Relative mtDNA copy number in vehicle treated (0.5% DMSO) 7dpf rrm2b^+/+ (n = 9)(circles with thin border) & rrm2b^−/− (n = 8)(squares with thin border) larvae and purine deoxynucleoside treated (50 μM dAdo & dGuo +5 μM ENHA in 0.5%DMSO) 7 dpf rrm2b^+/+ (n = 7)(circles with thick border) & rrm2b^−/− (n = 12)(squares with thick border) larvae. All results shown as mean ± SEM (statistical significance was determined with a Kruskal-Wallis test, ^*^*adj. P < 0.01). (B) Swim bladder area of 7 dpf vehicle treated (0.5% DMSO) rrm2b^+/+ (n = 19) (circles with thin border), rrm2b^+/− (n = 40) (diamonds), rrm2b^−/− (n = 18) (squares with thin border) and purine deoxynucleoside treated (50 μM dAdo & dGuo +5 μM ENHA in 0.5%DMSO) rrm2b^−/− larvae (n = 20) (squares with thick border). All results are shown as mean ± SEM (statistical significance was determined with a Kruskal-Wallis test, ^* = adj. P < 0.05, ^*^* = adj. P < 0.01, ^*^*^* = adj. p < 0.001). (C) L-lactate in 7 dpf vehicle treated (0.5% DMSO) rrm2b^+/+ & rrm2b^+/− larvae (circles with thin border) and rrm2b^−/− larvae (squares with thin border) and L-lactate in 7 dpf purine deoxynucleoside treated (50 μM dAdo & dGuo +5 μM ENHA in 0.5%DMSO) rrm2b^+/+ & rrm2b^+/− larvae (circles with thick border) and rrm2b^−/− larvae (squares with thick border). For all conditions n = 3 pools of 20, 7 dpf larvae. All results are shown as mean ± SEM (statistical significance was determined with a one-way ANOVA, ^* = adj. P < 0.05,^*^* = adj. P < 0.01). (D) Deoxyadenosine concentration (nmol/g protein) in pools (n = 80) of 7 dpf vehicle treated (0.5% DMSO) or purine deoxynucleoside treated (50 μM dAdo & dGuo +5 μM ENHA in 0.5%DMSO) rrm2b^+/+ & rrm2b^+/− and rrm2b^−/− larvae. E. Deoxyguanosine concentration (nmol/g protein) in pools (n = 80) of 7 dpf vehicle treated (0.5% DMSO) or purine deoxynucleoside treated (50 μM dAdo & dGuo +5 μM ENHA in 0.5%DMSO) rrm2b^+/+ & rrm2b^+/− and rrm2b^−/− larvae. Deoxycytidine monophosphate, dTMP: Deoxythymidine monophosphate, TK2: Thymidine kinase 2.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Phenotype Detail

Acknowledgments

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