SUR2-STOP zebrafish larvae exhibit increased seizure susceptibility and dysmorphology. (A) Automated swim tracking was used to measure motility in loss-of-function (LoF) SUR2-STOP fish and wild-type (WT) controls. Swimming distances prior to pentylenetetrazole (PTZ; 3 mM) administration (−) and after PTZ (+) shown. Data from individual measurements from biological replicates as dots with mean and standard error of the mean (SEM) shown. Measurements were made and combined from three separate breeding clutches. ***P < 0.001, ****P < 0.0001 from Tukey tests following one-way ANOVA. (B) Swimming distance for each larva after PTZ administration was normalized to basal activity prior to PTZ. Data from individual measurements from biological replicates shown as dots with mean and SEM shown. ****P < 0.0001 according to unpaired t-test. (C) Plot showing the cumulative distance swam for wild-type and SUR2-STOP larvae after PTZ administration. Swimming distances are normalized to the average swimming distances for each genotype over 30 s prior to PTZ admin. Data shown as mean (solid line) and SEM as shaded bars. (D–H) Morphometric analysis of wild-type and SUR2-STOP larvae showing reduced body length (D), equivalent head dimensions (E and F) and reduced inter-eye and eye diameter measurements (G and H) in SUR2-STOP larvae. Data from individual measurements from biological replicates as dots with mean and SEM shown. **P < 0.01 and ****P < 0.0001 according to unpaired t-tests.
|
