Evidence map›Paper›PMID 28374839›Full record

ArticleNature communications2017

Genetic silencing of olivocerebellar synapses causes dystonia-like behaviour in mice.

Joshua J White, Roy V Sillitoe

Open access · goldAbstract read
In one paragraph

Article in Nature communications, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 96 papers.

0numbers the graph read from it
0cells of the map it votes in
96citing papers in PubMed
10.5field-weighted citation impact, top 1% 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

96 citing papers in PubMed, 155 citations in OpenAlex.

  1. Article
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  11. Axonal pathology differentially affects human Purkinje cell subpopulations in the essential tremor cerebellum.Proceedings of the National Academy of Sciences of the United States of America · 2025
    Article
  12. Article
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  14. Thalamic deep brain stimulation improves movement in a cerebellar model of lesion-based status dystonicus.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2025
    Article
  15. Article
  16. Review
  17. Cerebellar deep brain stimulation as a dual-function therapeutic for restoring movement and sleep in dystonic mice.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2024
    Article
  18. Article
  19. Review
  20. Cerebellar Functions Beyond Movement and Learning.Annual review of neuroscience · 2024
    Review

36 more citing papers are in PubMed but not listed here.

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

2 authors at 2 institutions in 1 country.

Joshua J WhiteDepartment of Pathology and Immunology, Baylor College of Medicine, Houston, Texas 77030, USA.
Roy V SillitoeDepartment of Pathology and Immunology, Baylor College of Medicine, Houston, Texas 77030, USA.
Baylor College of Medicine · USTexas Children's Hospital · US

Funding

NRI Build-out: Advancing Translational Brain ResearchC06RR029965 · NCRR · BAYLOR COLLEGE OF MEDICINE · PI ZOGHBI, HUDA Y · 2010 to 2010
$14.9M
Steps towards a paternal gene activation therapy for Angelman syndromeU54HD083092 · NICHD · BAYLOR COLLEGE OF MEDICINE · PI NELSON, DAVID LOREN, NEUL, JEFFREY L · 2014 to 2019
$7.8M
SYNAPTIC ORIGINS OF CEREBELLAR DISEASER01NS089664 · NINDS · BAYLOR COLLEGE OF MEDICINE · PI SILLITOE, ROY VINCENT · 2014 to 2018
$1.7M
Tecan EVO GenePaint robotS10OD016167 · OD · BAYLOR COLLEGE OF MEDICINE · PI LJUNGBERG, CECILIA · 2013 to 2013
$215k
Cerebellar Function in DystoniaF31NS092264 · NINDS · BAYLOR COLLEGE OF MEDICINE · PI WHITE, JOSHUA J · 2015 to 2016
$69k
NCRR NIH HHS C06 RR029965NICHD NIH HHS U54 HD083092NIH HHS S10 OD016167NINDS NIH HHS F31 NS092264NINDS NIH HHS R01 NS089664
6 · The paper itself

Abstract

Theories of cerebellar function place the inferior olive to cerebellum connection at the centre of motor behaviour. One possible implication of this is that disruption of olivocerebellar signalling could play a major role in initiating motor disease. To test this, we devised a mouse genetics approach to silence glutamatergic signalling only at olivocerebellar synapses. The resulting mice had a severe neurological condition that mimicked the early-onset twisting, stiff limbs and tremor that is observed in dystonia, a debilitating movement disease. By blocking olivocerebellar excitatory neurotransmission, we eliminated Purkinje cell complex spikes and induced aberrant cerebellar nuclear activity. Pharmacologically inhibiting the erratic output of the cerebellar nuclei in the mutant mice improved movement. Furthermore, deep brain stimulation directed to the interposed cerebellar nuclei reduced dystonia-like postures in these mice. Collectively, our data uncover a neural mechanism by which olivocerebellar dysfunction promotes motor disease phenotypes and identify the cerebellar nuclei as a therapeutic target for surgical intervention.

Indexed as

Synaptic TransmissionAnimalsCerebellar NucleiCerebellumDeep Brain StimulationDisease Models, AnimalDystoniaGene Drive TechnologyGlutamic AcidMiceNeural PathwaysOlivary NucleusPurkinje CellsSynapsesTranscription FactorsVesicular Glutamate Transport Protein 2Glutamic AcidSlc17a6 protein, mousetranscription factor PTF1Transcription FactorsVesicular Glutamate Transport Protein 2

Identifiers

PMID28374839
PMCPMC5382291
OpenAlexW2605002006

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.