Evidence map›Paper›PMID 22532808›Full record

ArticlePLoS genetics2012

dyschronic, a Drosophila homolog of a deaf-blindness gene, regulates circadian output and Slowpoke channels.

James E C Jepson, Mohammad Shahidullah, Angelique Lamaze, Drew Peterson, Huihui Pan, Kyunghee Koh

Open access · goldAbstract read
In one paragraph

Article in PLoS genetics, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 22 papers.

0numbers the graph read from it
0cells of the map it votes in
22citing papers in PubMed
1.3field-weighted citation impact, top 24% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

22 citing papers in PubMed, 33 citations in OpenAlex.

  1. Article
  2. Review
  3. Review
  4. Cell-type-specific, multicolor labeling of endogenous proteins with split fluorescent protein tags inProceedings of the National Academy of Sciences of the United States of America · 2021
    Article
  5. Impaired Pre-Motor Circuit Activity and Movement in a Drosophila Model of KCNMA1-Linked Dyskinesia.Movement disorders : official journal of the Movement Disorder Society · 2021
    Article
  6. Article
  7. Article
  8. Article
  9. Article
  10. Review
  11. Review
  12. Article
  13. UsingDisease models & mechanisms · 2018
    Review
  14. Article
  15. Article
  16. Review
  17. The PDZ-domain protein Whirlin facilitates mechanosensory signaling in mammalian proprioceptors.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2015
    Article
  18. Article
  19. Cell-specific fine-tuning of neuronal excitability by differential expression of modulator protein isoforms.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2013
    Article
  20. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

6 authors at 3 institutions in 1 country.

James E C JepsonDepartment of Neuroscience, Farber Institute of Neuroscience, Kimmel Cancer Center, Thomas Jefferson University, Philadelphia, Pennsylvania, USA.
Mohammad Shahidullah
Angelique Lamaze
Drew Peterson
Huihui Pan
Kyunghee Koh
University of California, San Francisco · USSidney Kimmel Cancer Center · USThomas Jefferson University · US

Funding

X-Ray Crystallography and Macromolecular CharacterizationP30CA056036 · NCI · THOMAS JEFFERSON UNIVERSITY · PI DANIEL P SILVER · 1995 to 2026
$94.8M
REGULATION OF ION CHANNELS IN NERVE CELLSR01NS017910 · NINDS · UNIVERSITY OF PENNSYLVANIA · PI LEVITAN, IRWIN B · 1985 to 2009
$3.9M
Function of SLEEPLESS in sleep regulationR01GM088221 · NIGMS · UNIVERSITY OF PENNSYLVANIA · PI KOH, KYUNGHEE · 2009 to 2013
$1.4M
NCI NIH HHS P30 CA056036NCI NIH HHS P30CA56036NIGMS NIH HHS R01 GM088221NIGMS NIH HHS R01GM088221NINDS NIH HHS R01 NS017910NINDS NIH HHS R01NS017910
6 · The paper itself

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.

Indexed as

BrainDrosophila melanogasterNeuronsAnimalsBehavior, AnimalCircadian RhythmDeaf-Blind DisordersDrosophila ProteinsGene Expression RegulationHumansLarge-Conductance Calcium-Activated Potassium ChannelsMembrane ProteinsMotor ActivityProtein Interaction MapsClrn1 protein, ratDrosophila ProteinsLarge-Conductance Calcium-Activated Potassium ChannelsMembrane Proteinsslo protein, Drosophila

Identifiers

PMID22532808
PMCPMC3330124
OpenAlexW1978279909

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.