title: julius seizure, aDrosophilaMutant, Defines a Neuronal Population Underlying Epileptogenesis.
abstract: Epilepsy is a neural disorder characterized by recurrent seizures. Bang-sensitiveDrosophilarepresent an important model for studying epilepsy and neuronal excitability. Previous work identified the bang-sensitive geneslamdance(sda) as an allele of theaminopeptidase Ngene. Here we show through extensive genetic analysis, including recombination frequency, deficiency mapping, transposon insertion complementation testing, RNA interference (RNAi), and genetic rescue that the gene responsible for the seizure sensitivity isjulius seizure(jus), formerlyCG14509, which encodes a novel transmembrane domain protein. We also describe more severe genetic alleles ofjusRNAi-mediated knockdown ofjusrevealed that it is required only in neurons and not glia, and that partial bang-sensitivity is caused by knockdown in GABAergic or cholinergic but not glutamatergic neurons. RNAi knockdown ofjusat the early pupal stages leads to strong seizures in adult animals, implicating that stage as critical for epileptogenesis. A C-terminal-tagged version of Jus was generated from a fosmid genomic clone. This fosmid fusion rescued the bang-sensitive phenotype and was expressed in the optic lobes and the subesophageal and thoracic abdominal ganglia. The protein was primarily localized in axons, especially in the neck connectives, extending into the thoracic abdominal ganglion.
authors.affiliation: Department of Neurobiology and Behavior, Cornell University, Ithaca, New York 14853.
authors.forename: Meghan
authors.initials: M
authors.lastname: Horne
authors.affiliation: Department of Neurobiology and Behavior, Cornell University, Ithaca, New York 14853.
authors.forename: Kaitlyn
authors.initials: K
authors.lastname: Krebushevski
authors.affiliation: Department of Biomedical Engineering, Cornell University, Ithaca, New York 14853.
authors.forename: Amelia
authors.initials: A
authors.lastname: Wells
authors.affiliation: Weill Cornell Medicine-Qatar, Education City, Doha, Qatar.
authors.forename: Nahel
authors.initials: N
authors.lastname: Tunio
authors.affiliation: Department of Neurobiology and Behavior, Cornell University, Ithaca, New York 14853.
authors.forename: Casey
authors.initials: C
authors.lastname: Jarvis
authors.affiliation: Department of Neurobiology and Behavior, Cornell University, Ithaca, New York 14853.
authors.forename: Glen
authors.initials: G
authors.lastname: Francisco
authors.affiliation: Department of Neurobiology and Behavior, Cornell University, Ithaca, New York 14853.
authors.forename: Jane
authors.initials: J
authors.lastname: Geiss
authors.affiliation: Department of Neurobiology and Behavior, Cornell University, Ithaca, New York 14853.
authors.forename: Andrew
authors.initials: A
authors.lastname: Recknagel
authors.affiliation: Department of Neurobiology and Behavior, Cornell University, Ithaca, New York 14853 dld14@cornell.edu.
authors.forename: David L
authors.initials: DL
authors.lastname: Deitcher
mesh_terms: D000626:Aminopeptidases; D000818:Animals; D059329:Cholinergic Neurons / Q000378:metabolism* / Q000502:physiology; D004330:Drosophila / Q000166:cytology / Q000235:genetics* / Q000502:physiology; D029721:Drosophila Proteins / Q000235:genetics* / Q000378:metabolism; D059330:GABAergic Neurons / Q000378:metabolism* / Q000502:physiology; D017353:Gene Deletion; D008565:Membrane Proteins / Q000235:genetics* / Q000378:metabolism; D012640:Seizures / Q000235:genetics*
chemical_list: D029721:Drosophila Proteins; D008565:Membrane Proteins; C000609534:jus protein, Drosophila; D000626:Aminopeptidases
keywords: GABAergic; bang-sensitive; cholinergic; epilepsy; slamdance