FIGURE

Fig. 6

ID
ZDB-FIG-210611-34
Publication
Griffin et al., 2021 - Phenotypic analysis of catastrophic childhood epilepsy genes
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Fig. 6

Survival and behavioral phenotypes.

a Heatmap displaying median wild-type (WT), heterozygote (HET), and homozygote mutant (MUT) larval survival for EZP lines. Range extends from 8 dpf (red) to 13 dpf (blue). Asterisks indicate MUTs with significant survival deficits compared WT control siblings; p < 0.05, log rank test. b Lines with significant survival deficits. c Quantification of the basal locomotor activity of epileptic lines after 1 hr habituation in DanioVision chamber. Maximum velocity and total distance traveled were extracted directly from EthoVision XT 11.5 software while the number of events ≥28 mm/s, termed high-speed events (HSE), and long duration HSE (≥ 1 s) were scored using a MATLAB algorithm (scn1lab552 WT N = 19 larvae, MUT N = 31 larvae; scn1lab WT N = 21 larvae, MUT N = 16 larvae; arxa WT N = 25 larvae, MUT N = 22 larvae; strada WT N = 27 larvae, MUT N = 31 larvae; stxbp1b WT N = 26 larvae, MUT N = 43 larvae; pnpo WT N = 42 larvae, MUT N = 40 larvae; gabrb3 WT N = 35 larvae, MUT N = 36 larvae; eef1a2 WT N = 30 larvae, MUT N = 27 larvae and grin1b WT N = 29 larvae and HET = 57 larvae). d Representative traces of arxa WT and MUT movement. e Comparison of duration of HSE in scn1lab ENU and CRISPR larvae. Data displayed as mean ± SEM, one-way ANOVA was used to determine the significance of both HET and MUT behavior for all lines (Supplementary Figure 4). Post hoc Dunnett multiple comparison test, *p ≤ 0.05, **p ≤ 0.005, **p < 0.0001.

Expression Data

Expression Detail
Antibody Labeling
Phenotype Data
Fish:
Observed In:
Stage Range: Day 5 to Days 7-13

Phenotype Detail
Acknowledgments
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