Reference Report

Reference
Citation	Jepson, J.E., Shahidullah, M., Lamaze, A., Peterson, D., Pan, H., Koh, K. (2012). dyschronic, a Drosophila Homolog of a Deaf-Blindness Gene, Regulates Circadian Output and Slowpoke Channels. PLoS Genet. 8(4): e1002671. (Export to RIS)
FlyBase ID	FBrf0218124
Publication Type	Research paper
PubMed ID	22532808
PubMed Abstract	Many aspects of behavior and physiology are under circadian control. In Drosophila, the molecular clock that regulates rhythmic patterns of behavior has been extensively characterized. In contrast, genetic loci involved in linking the clock to alterations in motor activity have remained elusive. In a forward-genetic screen, we uncovered a new component of the circadian output pathway, which we have termed dyschronic (dysc). dysc mutants exhibit arrhythmic locomotor behavior, yet their eclosion rhythms are normal and clock protein cycling remains intact. Intriguingly, dysc is the closest Drosophila homolog of whirlin, a gene linked to type II Usher syndrome, the leading cause of deaf-blindness in humans. Whirlin and other Usher proteins are expressed in the mammalian central nervous system, yet their function in the CNS has not been investigated. We show that DYSC is expressed in major neuronal tracts and regulates expression of the calcium-activated potassium channel SLOWPOKE (SLO), an ion channel also required in the circadian output pathway. SLO and DYSC are co-localized in the brain and control each other's expression post-transcriptionally. Co-immunoprecipitation experiments demonstrate they form a complex, suggesting they regulate each other through protein-protein interaction. Furthermore, electrophysiological recordings of neurons in the adult brain show that SLO-dependent currents are greatly reduced in dysc mutants. Our work identifies a Drosophila homolog of a deaf-blindness gene as a new component of the circadian output pathway and an important regulator of ion channel expression, and suggests novel roles for Usher proteins in the mammalian nervous system.
DOI	10.1371/journal.pgen.1002671
Related Publication(s)
Recent Updates
Description	What does this section display? This section contains items that were added to this record for each release. It currently only tracks new links between this FlyBase report and other FlyBase data classes (e.g. genes, references, stocks) or controlled vocabulary terms (e.g. GO, anatomy terms). What does this section not display? This section does not currently display links that were removed or gene model changes.
Update Feed	Click the icon below to subscribe to this FlyBase record and receive updates automatically through your feed reader.
FB2013_03
FB2013_02
All updates	Click here to see a list of all updates to this record from FB2010_08 and on.
Associated Information
Comments
Associated Files
Other Information
Secondary IDs
Language of Publication	English
Additional Languages of Abstract
Also Published As
Parent Publication
Publication Type	Journal
Abbreviation	PLoS Genet.
Title	PLoS Genetics
Publication Year	2005-
ISBN/ISSN	1553-7404 1553-7390
Data from Reference
Aberrations (1)
	Df(3L)BSC614
Alleles (25)
	dysc^c03838 dysc^c05107 dysc^pr9e dysc^s168 dysc^Scer\UAS.f dysc^Scer\UAS.g Mmus\Cd8a^{Scer\UAS.T:Avic\GFP} Scer\GAL4^c061 Scer\GAL4^c107 Scer\GAL4^C164 Scer\GAL4^c819 Scer\GAL4^dimm-929 Scer\GAL4^dysc.PJ Scer\GAL4^elav.PU Scer\GAL4^ey-OK107 Scer\GAL4^how-24B Scer\GAL4^Ilp2.PR Scer\GAL4^P2.4.Pdf Scer\GAL4^repo.PU Scer\GAL4^Tab2-201Y Scer\GAL4^tim.PE Scer\GAL4^VGlut-OK371 Sh⁸ Sh²⁵ slo⁴
Constructs (10)
	P{dysc-GAL4.J} P{elav-GAL4.U} P{GAL4-tim.E} P{Ilp2-GAL4.R} P{Pdf-GAL4.P2.4} P{repo-GAL4.U} P{UAS-dysc.f} P{UAS-dysc.g} P{UAS-mCD8::GFP.L} P{XP}
Genes (7)
	dysc Mmus\Cd8a Pdf per Scer\GAL4 Sh slo
Insertions (10)
	P{GawB}c061 P{GawB}c107 P{GawB}C164 P{GawB}c819 P{GawB}dimm⁹²⁹ P{GawB}ey^OK107 P{GawB}how^24B P{GawB}Tab2^201Y P{GawB}VGlut^OK371 P{XP}dysc^s168
Natural transposons (1)
	P-element
Transcripts (2)
	Scer\GAL4^C164RA Scer\GAL4^dysc.PJRA