Figure S4.

Figure S4. PTZ and PTX treatments in cln3-deficient zebrafish larvae.

(A) Heterozygous and homozygous mutant cln3 larvae (5 dpf) were placed in individual wells of a flat-bottom 96-well plate. Subacute (5, 7.5, and 10 mM) and acute (15 and 20 mM) concentrations of PTZ were added, and 10 min after addition of the drug (habituation), the locomotor activity was tracked and the mean velocity was calculated for each condition (40 min). (B) Average distance moved within each 2-min time bin under 0 and 10 mM PTZ yields a similar behavioral profile for both genotypes, but higher activity was observed in MUT1 larvae. Error bars represent the SEM, plotted only below the means for the sake of clarity (n = 12). (C) Similar procedure was performed with PTX as described for PTZ. (D) Behavioral profile under PTX exposure (0.3 and 1 mM). Error bars represent SDs, plotted only below the means for the sake of clarity (n = 12 for each genotype). Statistically significant differences between lines were determined using the two-way ANOVA test followed by Dunnett’s multiple comparison test (****P ≤ 0.0001).