Evidence map›Paper›PMID 26586559›Full record

ArticleNeurobiology of disease2016

Pathogenesis of severe ataxia and tremor without the typical signs of neurodegeneration.

Joshua J White, Marife Arancillo, Annesha King, Tao Lin, Lauren N Miterko, Samrawit A Gebre, Roy V Sillitoe

Open access · greenAbstract read
In one paragraph

Article in Neurobiology of disease, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 31 papers.

0numbers the graph read from it
0cells of the map it votes in
31citing papers in PubMed
1.5field-weighted citation impact, top 19% 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

31 citing papers in PubMed, 59 citations in OpenAlex.

  1. Article
  2. Article
  3. Article
  4. Article
  5. Article
  6. Article
  7. Article
  8. Cerebellar α6GABANeurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2023
    Article
  9. Article
  10. Article
  11. Article
  12. Article
  13. Article
  14. Article
  15. Article
  16. DeletingeLife · 2021
    Article
  17. Article
  18. Article
  19. 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

7 authors at 2 institutions in 1 country.

Joshua J WhiteDepartment of Pathology & Immunology, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA; Department of Neuroscience, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA; Jan and Dan Duncan Neurological Research Institute of Texas Children's Hospital, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA.
Marife ArancilloDepartment of Pathology & Immunology, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA; Jan and Dan Duncan Neurological Research Institute of Texas Children's Hospital, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA.
Annesha KingDepartment of Pathology & Immunology, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA; Jan and Dan Duncan Neurological Research Institute of Texas Children's Hospital, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA.
Tao LinDepartment of Pathology & Immunology, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA; Jan and Dan Duncan Neurological Research Institute of Texas Children's Hospital, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA.
Lauren N MiterkoProgram in Developmental Biology, Baylor College of Medicine, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA; Jan and Dan Duncan Neurological Research Institute of Texas Children's Hospital, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA.
Samrawit A GebreDepartment of Pathology & Immunology, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA; Jan and Dan Duncan Neurological Research Institute of Texas Children's Hospital, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA.
Roy V SillitoeDepartment of Pathology & Immunology, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA; Department of Neuroscience, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA; Program in Developmental Biology, Baylor College of Medicine, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA; Jan and Dan Duncan Neurological Research Institute of Texas Children's Hospital, 1250 Moursund Street, Suite 1325, Houston, TX 77030, USA.
Shriners Hospitals for Children - Houston · USBaylor College of Medicine · US

Funding

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
YEARS 19 - 23: BCM POST-BACCALAUREATE RESEARCH EDUCATION PROGRAMR25GM069234 · NIGMS · BAYLOR COLLEGE OF MEDICINE · PI PEREIRA, FRED A · 2003 to 2024
$7.5M
SYNAPTIC ORIGINS OF CEREBELLAR DISEASER01NS089664 · NINDS · BAYLOR COLLEGE OF MEDICINE · PI SILLITOE, ROY VINCENT · 2014 to 2018
$1.7M
Cerebellar Function in DystoniaF31NS092264 · NINDS · BAYLOR COLLEGE OF MEDICINE · PI WHITE, JOSHUA J · 2015 to 2016
$69k
NICHD NIH HHS U54 HD083092NICHD NIH HHS U54HD083092NIGMS NIH HHS R25 GM069234NINDS NIH HHS F31 NS092264NINDS NIH HHS F31NS092264NINDS NIH HHS R01 NS089664
6 · The paper itself

Abstract

Neurological diseases are especially devastating when they involve neurodegeneration. Neuronal destruction is widespread in cognitive disorders such as Alzheimer's and regionally localized in motor disorders such as Parkinson's, Huntington's, and ataxia. But, surprisingly, the onset and progression of these diseases can occur without neurodegeneration. To understand the origins of diseases that do not have an obvious neuropathology, we tested how loss of CAR8, a regulator of IP3R1-mediated Ca(2+)-signaling, influences cerebellar circuit formation and neural function as movement deteriorates. We found that faulty molecular patterning, which shapes functional circuits called zones, leads to alterations in cerebellar wiring and Purkinje cell activity, but not to degeneration. Rescuing Purkinje cell function improved movement and reducing their Ca(2+) influx eliminated ectopic zones. Our findings in Car8(wdl) mutant mice unveil a pathophysiological mechanism that may operate broadly to impact motor and non-motor conditions that do not involve degeneration.

Indexed as

AnimalsAtaxiaBiomarkers, TumorCarbonic AnhydrasesCerebellumChlorzoxazoneLearningMiceMice, Inbred C57BLMotor ActivityNerve Tissue ProteinsNeural PathwaysPurkinje CellsSpinal CordTremorTyrosine 3-MonooxygenaseBiomarkers, TumorCar8 protein, mouseCarbonic AnhydrasesChlorzoxazoneNerve Tissue ProteinsTyrosine 3-MonooxygenaseAtaxiaBehaviorCerebellumIn vivo electrophysiologyNeurodegenerationTremor

Identifiers

PMID26586559
PMCPMC4778569
OpenAlexW2179205971

What OpenQuestion holds

Textmetadata
LicenceTDM
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.