Figure 1

Characterization of epileptic phenotype in stxbp1b mutant zebrafish

(A) Representative spontaneous high-velocity convulsive behavior captured during high-speed imaging (250 fps) of a single stxbp1b mutant larva freely swimming in embryo media at room temperature. Note these were rare events that occur with a velocity near, or greater than, the acquisition speed of the camera (QImaging Optimos cMOS).

(B) Representative 15 min local field potential recordings from randomly selected larvae from a cross of stxbp1b^+/- adult breeders. LFP recordings were obtained from a glass microelectrode positioned under a microscope in the midbrain of agarose-embedded larvae at 5 dpf. Larvae were freed from the agarose and genotyped post hoc. Note the presence of small amplitude events building to a large amplitude multi-spike ictal event with postictal depression in the stxbp1b mutant larvae (see inset) but not the WT sibling. These recordings are representative of the spontaneous and unprovoked seizure activity associated with this zebrafish line.

(C) Confocal images taken with a 2-photon microscope of a representative neurod1:GCaMP6f expressing larval zebrafish at three different levels through the central nervous system. Scale bar, 100 μm