Evidence map›Paper›PMID 33300869›Full record

ArticleeLife2020

Vanishing white matter disease expression of truncated EIF2B5 activates induced stress response.

Matthew D Keefe, Haille E Soderholm, Hung-Yu Shih, Tamara J Stevenson, Kathryn A Glaittli, D Miranda Bowles, Erika Scholl, Samuel Colby, Samer Merchant, Edward W Hsu and 1 more

Open access · goldAbstract read
In one paragraph

Article in eLife, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers.

0numbers the graph read from it
0cells of the map it votes in
15citing papers in PubMed
1.0field-weighted citation impact, top 25% 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

15 citing papers in PubMed, 23 citations in OpenAlex.

  1. Male germ cell-specific deletion ofZoological research · 2026
    Article
  2. Review
  3. Review
  4. Evaluation of safety and early efficacy of AAV gene therapy in mouse models of vanishing white matter disease.Molecular therapy : the journal of the American Society of Gene Therapy · 2024
    Article
  5. Review
  6. Article
  7. Article
  8. Review
  9. Disease Modeling of Rare Neurological Disorders in Zebrafish.International journal of molecular sciences · 2022
    Review
  10. Review
  11. Article
  12. Review
  13. Review
  14. Article
  15. Proteome Profile of Myelin in the Zebrafish Brain.Frontiers in cell and developmental biology · 2021
    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

11 authors at 2 institutions in 1 country.

Matthew D Keefe *Department of Pediatrics, University of Utah School of Medicine, Salt Lake City, United States.
Haille E Soderholm *Department of Pediatrics, University of Utah School of Medicine, Salt Lake City, United States.
Hung-Yu Shih *Department of Pediatrics, University of Utah School of Medicine, Salt Lake City, United States.
Tamara J StevensonDepartment of Pediatrics, University of Utah School of Medicine, Salt Lake City, United States.
Kathryn A GlaittliDepartment of Pediatrics, University of Utah School of Medicine, Salt Lake City, United States.
D Miranda BowlesDepartment of Pediatrics, University of Utah School of Medicine, Salt Lake City, United States.
Erika SchollDepartment of Pediatrics, University of Utah School of Medicine, Salt Lake City, United States.
Samuel ColbyDepartment of Bioengineering, University of Utah, Salt Lake City, United States.
Samer MerchantDepartment of Bioengineering, University of Utah, Salt Lake City, United States.
Edward W HsuDepartment of Bioengineering, University of Utah, Salt Lake City, United States.
Joshua L BonkowskyDepartment of Pediatrics, University of Utah School of Medicine, Salt Lake City, United States.ORCID 0000-0001-8775-147X
University of Utah · USPrimary Children's Hospital · US

Funding

Utah Center for Clinical and Translational ScienceUL1TR002538 · NCATS · UNIVERSITY OF UTAH · PI HESS, RACHEL, MAJERSIK, JENNIFER JUHL · 2018 to 2022
$26.0M
Development and Validation of a Zebrafish Model for Vanishing White Matter DiseaseR21NS109441 · NINDS · UNIVERSITY OF UTAH · PI BONKOWSKY, JOSH LEITCH · 2019 to 2020
$753k
Expansion of a Zebrafish Research Core FacilityG20OD018369 · OD · UNIVERSITY OF UTAH · PI GRUNWALD, DAVID J. · 2014 to 2014
$500k
NCRR Confocal ProposalS10RR024761 · NCRR · UNIVERSITY OF UTAH · PI RODESCH, CHRISTOPHER K · 2008 to 2008
$475k
NCATS NIH HHS UL1 TR002538NCRR NIH HHS S10 RR024761NIH HHS 1R21 NS109441-01NIH HHS G20 OD018369NINDS NIH HHS R21 NS109441
6 · The paper itself

Abstract

Vanishing white matter disease (VWM) is a severe leukodystrophy of the central nervous system caused by mutations in subunits of the eukaryotic initiation factor 2B complex (eIF2B). Current models only partially recapitulate key disease features, and pathophysiology is poorly understood. Through development and validation of zebrafish (

Indexed as

Disease Models, AnimalAnimalsEukaryotic Initiation Factor-2BHumansLeukoencephalopathiesMutationStress, PhysiologicalZebrafishEukaryotic Initiation Factor-2BEIF2B5induced stress responseleukodystrophymedicineMRImyelinvanishing white matter diseasezebrafish

Identifiers

PMID33300869
PMCPMC7752137
OpenAlexW3111778161

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.