Spontaneous seizures and altered gene expression in GABA signaling pathways in a mind bomb mutant zebrafish.

ABSTRACT: Disruption of E3 ubiquitin ligase activity in immature zebrafish mind bomb mutants leads to a failure in Notch signaling, excessive numbers of neurons, and depletion of neural progenitor cells. This neurogenic phenotype is associated with defects in neural patterning and brain development. Because developmental brain abnormalities are recognized as an important feature of childhood neurological disorders such as epilepsy and autism, we determined whether zebrafish mutants with grossly abnormal brain structure exhibit spontaneous electrical activity that resembles the long-duration, high-amplitude multispike discharges reported in immature zebrafish exposed to convulsant drugs. Electrophysiological recordings from agar immobilized mind bomb mutants at 3 d postfertilization confirmed the occ

SUBMITTER: Hortopan GA 

PROVIDER: S-EPMC2962868 | biostudies-literature | 2010 Oct

REPOSITORIES: biostudies-literature