FIGURE

Fig 2

ID

ZDB-FIG-200316-21

Publication

Weuring et al., 2020 - NaV1.1 and NaV1.6 selective compounds reduce the behavior phenotype and epileptiform activity in a novel zebrafish model for Dravet Syndrome

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Fig 2

A) Spontaneous burst movements quantified in two knockout lines using the locomotor burst movement assay B) Burst movements are unique to <italic>Scn1lab</italic> knockouts, and not caused by the absence of an inflated swimming bladder C) The burst movement phenotype of <italic>Scn1lab</italic> knockouts is partially rescued when human SCN1A, either mRNA or cDNA is introduced D) <italic>Scn1Lab</italic> knockouts show a reduction in free GABA levels E) Both heterozygote and homozygous <italic>Scn1lab</italic> knockouts show sensitivity to exposure of 5mM pentylenetetrazole F) Representative non-invasive local field potential recordings from the brain of wildtype and <italic>Scn1Lab</italic> knockout zebrafish (n = 3).

Only two types of signals can be detected in wildtype embryos: low amplitude waves and very occasional sharp single spikes from an otherwise straight and silent baseline (1 and 2). In Scn1Lab knockouts, at least three types of signal differ from wildtype recordings including trains of biphasic spikes lasting seconds (3) and several short spike events (4 and 5) which resembles epileptiform activity. Error bars = S.D. (-/-) = Scn1Lab knockout, locomotor assay n = 12 per group * <0.05 **<0.005 ***<0.0005.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	WT scn1lab^zu1/zu1 scn1lab^zu1/+
Condition:	chemical treatment by environment: pentetrazol
Observed In:	brain neuronal action potential swimming whole organism gamma-aminobutyric acid
Stage:	Day 5

Phenotype Detail

Acknowledgments

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