ZFIN Image: Butti et al., 2021, Fig. 2

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Figure Caption

Fig. 2 Motor behavioural deficits and reduced acetylcholine receptor clusters at neuromuscular junctions (NMJs) in C9-miR fish.

a Representative swimming tracks of control, C9-miR and C9-rescue fish at 6 dpf. Scale bar = 0.2 cm. b C9-miR larvae (N = 3, n = 65) displayed impaired swimming compared to controls (N = 3, n = 60; ***p < 0.0001; one-way ANOVA). Expression of the human C9orf72 long transcript in C9-miR fish (C9-rescue; N = 3, n = 53) significantly rescued the motor behavioural defects (***p < 0.0001; one-way ANOVA). c Representative images of co-immunostaining of zebrafish neuromuscular junctions with presynaptic (SV2; green) and postsynaptic (α-bungarotoxin; red) markers in 6 dpf zebrafish. Scale bar = 100 μm. d Quantification of the colocalizing presynaptic and postsynaptic markers per somite showed a significant reduction in the number of puncta in C9-miR fish at 6 dpf that can be rescued with the expression of human C9orf72 mRNA (C9-rescue) (n = 8–10; ***p < 0.0001; one-way ANOVA). Data are presented as mean ± SEM. n represents the number of fish, N represents the number of experimental repeats.

Acknowledgments

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