Evidence map›Paper›PMID 37043392›Full record

ArticleFASEB journal : official publication of the Federation of American Societies for Experimental Biology2023

Gigaxonin is required for intermediate filament transport.

Bhuvanasundar Renganathan, James P Zewe, Yuan Cheng, Jean-Michel Paumier, Mark Kittisopikul, Karen M Ridge, Puneet Opal, Vladimir I Gelfand

Open access · hybridAbstract read
In one paragraph

Article in FASEB journal : official publication of the Federation of American Societies for Experimental Biology, 2023. 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
3.8field-weighted citation impact, top 7% 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, 18 citations in OpenAlex.

  1. Article
  2. Article
  3. Vimentin Intermediate Filaments: A Paradigm Shift From Static Structure to Dynamic Cytoplasmic Network.BioEssays : news and reviews in molecular, cellular and developmental biology · 2026
    Review
  4. Gigaxonin Potentiates Antiviral Innate Immune Responses by Targeting cGAS and TREX1.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2025
    Article
  5. Article
  6. Article
  7. Article
  8. Article
  9. Ubiquitin system mutations in neurological diseases.Trends in biochemical sciences · 2024
    Review
  10. Article
  11. Article
  12. Review
  13. Review
  14. Gigaxonin is required for intermediate filament transport.FASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2023
    Article
  15. Neurofilaments in health and Charcot-Marie-Tooth disease.Frontiers in cell and developmental biology · 2023
    Review
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

8 authors at 1 institution in 1 country.

Bhuvanasundar RenganathanDepartment of Cell and Developmental Biology, Feinberg School of Medicine, Northwestern University, Chicago, Illinois, USA.ORCID https://orcid.org/0000-0003-1878-3739
James P ZeweKen and Ruth Davee Department of Neurology, Feinberg School of Medicine, Northwestern University, Chicago, Illinois, USA.
Yuan ChengDivision of Pulmonary and Critical Care Medicine, Department of Medicine, Northwestern University, Feinberg School of Medicine, Chicago, Illinois, USA.
Jean-Michel PaumierKen and Ruth Davee Department of Neurology, Feinberg School of Medicine, Northwestern University, Chicago, Illinois, USA.
Mark KittisopikulDepartment of Cell and Developmental Biology, Feinberg School of Medicine, Northwestern University, Chicago, Illinois, USA.
Karen M RidgeDivision of Pulmonary and Critical Care Medicine, Department of Medicine, Northwestern University, Feinberg School of Medicine, Chicago, Illinois, USA.
Puneet OpalKen and Ruth Davee Department of Neurology, Feinberg School of Medicine, Northwestern University, Chicago, Illinois, USA.
Vladimir I GelfandDepartment of Cell and Developmental Biology, Feinberg School of Medicine, Northwestern University, Chicago, Illinois, USA.ORCID https://orcid.org/0000-0002-6361-2798
Northwestern University · US

Funding

Tumor Environment and Metastasis (TEAM) Research ProgramP30CA060553 · NCI · NORTHWESTERN UNIVERSITY AT CHICAGO · PI Daniela E Matei · 1993 to 2026
$153.9M
Structural basis of the specific protein-protein interactions underlying IF assemP01GM096971 · NIGMS · NORTHWESTERN UNIVERSITY AT CHICAGO · PI GOLDMAN, ROBERT D · 2011 to 2021
$16.8M
Microtubule motors, cytoskeletal organization and cell polarityR35GM131752 · NIGMS · NORTHWESTERN UNIVERSITY AT CHICAGO · PI Vladimir I Gelfand · 2019 to 2026
$6.4M
Understanding the Cellular Basis of Movement DisordersR01NS082351 · NINDS · NORTHWESTERN UNIVERSITY AT CHICAGO · PI Puneet Opal · 2013 to 2026
$6.2M
Elucidating cellular mechanisms underlying neurodegenerationR01NS127204 · NINDS · NORTHWESTERN UNIVERSITY AT CHICAGO · PI Puneet Opal · 2022 to 2026
$2.8M
NCI NIH HHS P30 CA060553NIGMS NIH HHS P01 GM096971NIGMS NIH HHS R35 GM131752NINDS NIH HHS R01 NS082351NINDS NIH HHS R01 NS127204
6 · The paper itself

Abstract

Gigaxonin is an adaptor protein for E3 ubiquitin ligase substrates. It is necessary for ubiquitination and degradation of intermediate filament (IF) proteins. Giant axonal neuropathy is a pathological condition caused by mutations in the GAN gene that encodes gigaxonin. This condition is characterized by abnormal accumulation of IFs in both neuronal and non-neuronal cells; however, it is unclear what causes IF aggregation. In this work, we studied the dynamics of IFs using their subunits tagged with a photoconvertible protein mEOS 3.2. We have demonstrated that the loss of gigaxonin dramatically inhibited transport of IFs along microtubules by the microtubule motor kinesin-1. This inhibition was specific for IFs, as other kinesin-1 cargoes, with the exception of mitochondria, were transported normally. Abnormal distribution of IFs in the cytoplasm can be rescued by direct binding of kinesin-1 to IFs, demonstrating that transport inhibition is the primary cause for the abnormal IF distribution. Another effect of gigaxonin loss was a more than 20-fold increase in the amount of soluble vimentin oligomers in the cytosol of gigaxonin knock-out cells. We speculate that these oligomers saturate a yet unidentified adapter that is required for kinesin-1 binding to IFs, which might inhibit IF transport along microtubules causing their abnormal accumulation.

Indexed as

Cytoskeletal ProteinsGiant Axonal NeuropathyHumansIntermediate Filament ProteinsIntermediate FilamentsKinesinsMicrotubulesCytoskeletal ProteinsIntermediate Filament ProteinsKinesinsgiant axonal neuropathygigaxoninintermediate filamentskinesin-1microtubulesneurofilamentsvimentin

Identifiers

PMID37043392
PMCPMC10237250
OpenAlexW4365135670

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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.