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Fig. 5 | Genome Medicine

Fig. 5

From: Functional characterisation of the amyotrophic lateral sclerosis risk locus GPX3/TNIP1

Fig. 5

Knockdown of gpx3 in zebrafish-MO results in a motor phenotype. A Zebrafish embryos were injected at the one-cell stage with or without control MO (Control-MO), anti-gpx3 MO (gpx3-MO) or combined anti-gpx3 MO with 100 pg of cst-gpx3 mRNA (gpx3-rescue). Morphological and behavioural analyses were carried out post-fertilisation. B A range of MO concentrations targeting gpx3 indicated a dose-dependent motor-phenotype effect, which peaked at 1 mM without developmental abnormalities (age: 2dpf). C–E The gpx3-MO (1 mM) injected animals moved a smaller distance, for fewer minutes and with an overall lower speed compared to both un-injected and CTR-MO-injected controls. The motor defects were all significantly rescued following co-injection with MO-insensitive gpx3-mRNA (cst-gpx3) (n = 67–158, mean ± 95% CI). Significance indicates the comparison between control-MO and gpx3-MO (p < 0.001). Graphs show individual values, mean and 95% CI error bars. Swim distance, time and speed mean difference and 95% CI: 112 ± 28 mm, 1.29 ± 0.59 s and 32.0 ± 2.53 mm/s, respectively

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