Evidence map›Paper›PMID 28389476›Full record

ArticleThe Journal of cell biology2017

Defects in ER-endosome contacts impact lysosome function in hereditary spastic paraplegia.

Rachel Allison, James R Edgar, Guy Pearson, Tania Rizo, Timothy Newton, Sven Günther, Fiamma Berner, Jennifer Hague, James W Connell, Jürgen Winkler and 4 more

Open access · hybridAbstract read
In one paragraph

Article in The Journal of cell biology, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 110 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
110citing papers in PubMed, 2 pooled it
10.1field-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

110 citing papers in PubMed, 2 syntheses or guidelines pooled it, 168 citations in OpenAlex.

  1. Stay in touch with the endoplasmic reticulum.Science China. Life sciences · 2024
    Pooled it
  2. Frontiers in neuroscience · 2019
    Pooled it
  3. Review
  4. Article
  5. Review
  6. Review
  7. ESCRT-III function in membrane fission and repair.Nature reviews. Molecular cell biology · 2026
    Review
  8. Article
  9. Article
  10. Article
  11. Article
  12. Review
  13. Article
  14. Unraveling Isoform Complexity: The Roles of M1- and M87-Spastin in Spastic Paraplegia 4 (SPG4).Movement disorders : official journal of the Movement Disorder Society · 2025
    Review
  15. The Endoplasmic Reticulum and Its Contacts: Emerging Roles in Axon Development, Neurotransmission, and Degeneration.The Neuroscientist : a review journal bringing neurobiology, neurology and psychiatry · 2024
    Review
  16. Article
  17. Three-dimensional architecture of ESCRT-III flat spirals on the membrane.Proceedings of the National Academy of Sciences of the United States of America · 2024
    Article
  18. Article
  19. A chemical inhibitor of IST1-CHMP1B interaction impairs endosomal recycling and induces noncanonical LC3 lipidation.Proceedings of the National Academy of Sciences of the United States of America · 2024
    Article
  20. Article

50 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

14 authors at 5 institutions in 3 countries.

Rachel AllisonCambridge Institute for Medical Research, University of Cambridge, Cambridge CB2 0XY, England, UK.ORCID 0000-0002-2446-8455
James R EdgarCambridge Institute for Medical Research, University of Cambridge, Cambridge CB2 0XY, England, UK.ORCID 0000-0001-7903-8199
Guy PearsonCambridge Institute for Medical Research, University of Cambridge, Cambridge CB2 0XY, England, UK.ORCID 0000-0001-8623-9239
Tania RizoInterdisciplinary Center for Clinical Research (IZKF) Junior Research Group III and Federal Ministry of Education and Research (BMBF) Research Group Neuroscience, Friedrich-Alexander-University Erlangen-Nuernberg, 91054 Erlangen, Germany.ORCID 0000-0003-4922-7719
Timothy NewtonCambridge Institute for Medical Research, University of Cambridge, Cambridge CB2 0XY, England, UK.
Sven GüntherDepartment of Clinical Chemistry and Laboratory Diagnostics, Jena University Hospital, 07743 Jena, Germany.ORCID 0000-0003-1118-8726
Fiamma BernerCambridge Institute for Medical Research, University of Cambridge, Cambridge CB2 0XY, England, UK.ORCID 0000-0001-5625-5537
Jennifer HagueCambridge Institute for Medical Research, University of Cambridge, Cambridge CB2 0XY, England, UK.ORCID 0000-0002-9779-8009
James W ConnellCambridge Institute for Medical Research, University of Cambridge, Cambridge CB2 0XY, England, UK.
Jürgen WinklerDepartment of Molecular Neurology, Friedrich-Alexander-University Erlangen-Nuernberg, 91054 Erlangen, Germany.ORCID 0000-0003-0630-9204
Jennifer Lippincott-SchwartzJanelia Research Campus, Howard Hughes Medical Institute, Ashburn, VA 20147.ORCID 0000-0002-8601-3501
Christian BeetzDepartment of Clinical Chemistry and Laboratory Diagnostics, Jena University Hospital, 07743 Jena, Germany.ORCID 0000-0001-7061-2895
Beate WinnerInterdisciplinary Center for Clinical Research (IZKF) Junior Research Group III and Federal Ministry of Education and Research (BMBF) Research Group Neuroscience, Friedrich-Alexander-University Erlangen-Nuernberg, 91054 Erlangen, Germany.
Evan ReidCambridge Institute for Medical Research, University of Cambridge, Cambridge CB2 0XY, England, UK ealr4@cam.ac.uk.ORCID 0000-0003-1623-7304
University of Cambridge · GBFriedrich-Alexander-Universität Erlangen-Nürnberg · DEJena University Hospital · DEBundesministerium für Forschung, Technologie und Raumfahrt · DEHoward Hughes Medical Institute · US

Funding

Medical Research Council MR/M00046X/1Wellcome Trust 082381Wellcome Trust 086598Wellcome Trust 093026Wellcome Trust 100140
6 · The paper itself

Abstract

Contacts between endosomes and the endoplasmic reticulum (ER) promote endosomal tubule fission, but the mechanisms involved and consequences of tubule fission failure are incompletely understood. We found that interaction between the microtubule-severing enzyme spastin and the ESCRT protein IST1 at ER-endosome contacts drives endosomal tubule fission. Failure of fission caused defective sorting of mannose 6-phosphate receptor, with consequently disrupted lysosomal enzyme trafficking and abnormal lysosomal morphology, including in mouse primary neurons and human stem cell-derived neurons. Consistent with a role for ER-mediated endosomal tubule fission in lysosome function, similar lysosomal abnormalities were seen in cellular models lacking the WASH complex component strumpellin or the ER morphogen REEP1. Mutations in

Indexed as

AdultAnimalsCells, CulturedEndoplasmic ReticulumEndosomesFemaleHeLa CellsHumansLysosomesMaleMiceMiddle AgedSpastic Paraplegia, Hereditary

Identifiers

PMID28389476
PMCPMC5412567
OpenAlexW2605245547

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.