FIGURE

Fig. 3

ID
ZDB-FIG-181214-3
Publication
Samarut et al., 2018 - γ-Aminobutyric acid receptor alpha 1 subunit loss of function causes genetic generalized epilepsy by impairing inhibitory network neurodevelopment
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Fig. 3

Generalized neuronal activity in gabra1−/− following light exposure and sudden death following generalized seizures. A, Gabra1+/− and NeuroD:GCaMP6f +/− were intercrossed, and neuronal activity was monitored under a confocal microscope at 8 days postfertilization. After a period of 30 minutes in complete darkness, switching on the laser induced a broad neuronal activity in both optic tecta in gabra1−/− (n = 12) compared to gabra1+/+ siblings (n = 10) that lasted for few seconds (arrows). The intense neuronal activity of −/− larvae was rescued to wild‐type (WT) level after exposure to valproic acid (VPA; 50 μmol/L, n = 7). B, Fluorescence quantification in the optic tecta before and after laser exposure shows a significant increase of fluorescence in −/− larvae compared to +/+ siblings. Each point corresponds to the relative quantification of fluorescence from a frame with 400‐millisecond exposure. VPA treatment fully rescues the fluorescence back to the WT level (Student t test: *P < 0.05, ** P < 0.01, ***P < 0.005). C, The whirlpool onset (ie, clonic‐like phase) increased with age, with a full penetrance at 6 weeks postfertilization (wpf) onward. D, Sudden unexpected death in epilepsy (SUDEP) events were quantified and occurred from 4 wpf onward only in gabra1−/− mutants. Of note is that data shown here correspond to the actual SUDEP events witnessed, but it is very likely that it occurs in many more fish

Expression Data

Expression Detail
Antibody Labeling
Phenotype Data
Fish:
Condition:
Observed In:
Stage Range: Days 7-13 to Days 45-89

Phenotype Detail
Acknowledgments
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