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Pizotifen malate increases total swimming activity and partially rescues locomotor deficits in C9orf72 knockout larvae. (A) Mean swimming distance per minute for pizotifen malate 0.5 µM (PM) treated (+) and untreated (-) C9orf72+/+, C9orf72-/+, and C9orf72-/- six day post-fertilization (dpf) zebrafish larvae under a 120-minute phasic dark-light program. Data are presented as mean ± SEM. (B) Quantitative analysis of total swimming distance shows significant increases in PM-treated (+) C9orf72+/+ and C9orf72-/- larvae compared to untreated (-) controls. Data are presented as mean ± SEM. Welch and Brown-Forsythe ANOVA with Dunnett#39;s T3 multiple comparisons post-hoc test. **** p < 0.0001, ** p ≤ 0.01, NS p > 0.05. Boxplot extremities indicate minimum and maximum values; box limits represent the central 50% of the data, and the central line marks the median. (C-E) Swimming distance per minute and representative 60-second swimming tracks for treated and untreated C9orf72+/+(C), C9orf72-/+(D), and C9orf72-/-(E) larvae. Data are presented as mean ± SEM. N = 5, experimental repeats; n = 80, larvae per genotype. Scale bars = 0.2 cm.
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