Open Close
Reference
Citation
Saras, A., Tanouye, M.A. (2016). Mutations of the Calcium Channel Gene cacophony Suppress Seizures in Drosophila.  PLoS Genet. 12(1): e1005784.
FlyBase ID
FBrf0230696
Publication Type
Research paper
Abstract

Bang sensitive (BS) Drosophila mutants display characteristic seizure-like phenotypes resembling, in some aspects, those of human seizure disorders such as epilepsy. The BS mutant parabss1, caused by a gain-of-function mutation of the voltage-gated Na+ channel gene, is extremely seizure-sensitive with phenotypes that have proven difficult to ameliorate by anti-epileptic drug feeding or by seizure-suppressor mutation. It has been presented as a model for intractable human epilepsy. Here we show that cacophony (cacTS2), a mutation of the Drosophila presynaptic Ca++ channel α1 subunit gene, is a particularly potent seizure-suppressor mutation, reverting seizure-like phenotypes for parabss1 and other BS mutants. Seizure-like phenotypes for parabss1 may be suppressed by as much as 90% in double mutant combinations with cacTS2. Unexpectedly, we find that parabss1 also reciprocally suppresses cacTS2 seizure-like phenotypes. The cacTS2 mutant displays these seizure-like behaviors and spontaneous high-frequency action potential firing transiently after exposure to high temperature. We find that this seizure-like behavior in cacTS2 is ameliorated by 85% in double mutant combinations with parabss1.

PubMed ID
PubMed Central ID
PMC4714812 (PMC) (EuropePMC)
Related Publication(s)
Erratum

Correction: Mutations of the Calcium Channel Gene cacophony Suppress Seizures in Drosophila.
PLOS Genetics Staff, 2016, PLoS Genet. 12(2): e1005871 [FBrf0230955]

Associated Information
Comments
Associated Files
Other Information
Secondary IDs
    Language of Publication
    English
    Additional Languages of Abstract
    Parent Publication
    Publication Type
    Journal
    Abbreviation
    PLoS Genet.
    Title
    PLoS Genetics
    Publication Year
    2005-
    ISBN/ISSN
    1553-7404 1553-7390
    Data From Reference
    Alleles (8)
    Gene Groups (1)
    Genes (5)
    Human Disease Models (3)
    Insertions (1)
    Transgenic Constructs (3)