Evidence map›Paper›PMID 31231042›Full record

ArticleDevelopmental cell2019

Lysosomal Regulation of Inter-mitochondrial Contact Fate and Motility in Charcot-Marie-Tooth Type 2.

Yvette C Wong, Wesley Peng, Dimitri Krainc

Open access · bronzeAbstract read
In one paragraph

Article in Developmental cell, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 60 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
60citing papers in PubMed, 2 pooled it
4.2field-weighted citation impact, top 5% 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

60 citing papers in PubMed, 2 syntheses or guidelines pooled it, 99 citations in OpenAlex.

  1. Pooled it
  2. Guideline
  3. Review
  4. Article
  5. Dysregulation of autophagosome-mitochondria contacts contributes to autophagy dysfunction and neurodegeneration in tauopathy.Proceedings of the National Academy of Sciences of the United States of America · 2026
    Article
  6. Review
  7. Review
  8. Article
  9. Article
  10. Review
  11. Article
  12. Molecular mechanisms of mitochondrial dynamics.Nature reviews. Molecular cell biology · 2025
    Review
  13. Article
  14. Mitochondrial dysfunction, cause or consequence in neurodegenerative diseases?BioEssays : news and reviews in molecular, cellular and developmental biology · 2025
    Review
  15. Article
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4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

3 authors at 1 institution in 1 country.

Yvette C WongDepartment of Neurology, Northwestern University Feinberg School of Medicine, Chicago, IL 60611, USA.
Wesley PengDepartment of Neurology, Northwestern University Feinberg School of Medicine, Chicago, IL 60611, USA.
Dimitri KraincDepartment of Neurology, Northwestern University Feinberg School of Medicine, Chicago, IL 60611, USA. Electronic address: dkrainc@nm.org.
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
General Motor Control Mechanisms and Disease Training ProgramT32NS041234 · NINDS · NORTHWESTERN UNIVERSITY AT CHICAGO · PI BEVAN, MARK D · 2001 to 2022
$6.0M
The Role of Lysosomal Glucocerebrosidase in SynucleinopathiesR01NS076054 · NINDS · NORTHWESTERN UNIVERSITY AT CHICAGO · PI KRAINC, DIMITRI · 2011 to 2020
$4.1M
Nikon Super-Resolution Structured Illumination Microscope (SIM)S10OD016342 · OD · NORTHWESTERN UNIVERSITY AT CHICAGO · PI CHEW, TENG-LEONG · 2013 to 2013
$600k
Molecular mechanisms underlying mitochondria-lysosome membrane contact sites in neuronal function and neurodegenerationK99NS109252 · NINDS · NORTHWESTERN UNIVERSITY AT CHICAGO · PI WONG, YVETTE · 2018 to 2019
$179k
NRSA Postdoctoral F32 Misregulation of ER membrane dynamics by alpha-synuclein in Parkinsons diseaseF32NS101778 · NINDS · NORTHWESTERN UNIVERSITY AT CHICAGO · PI WONG, YVETTE · 2017 to 2018
$76k
NCI NIH HHS P30 CA060553NIH HHS S10 OD016342NINDS NIH HHS F32 NS101778NINDS NIH HHS K99 NS109252NINDS NIH HHS R01 NS076054NINDS NIH HHS T32 NS041234
6 · The paper itself

Abstract

Properly regulated mitochondrial networks are essential for cellular function and implicated in multiple diseases. Mitochondria undergo fission and fusion events, but the dynamics and regulation of a third event of inter-mitochondrial contact formation remain unclear. Using super-resolution imaging, we demonstrate that inter-mitochondrial contacts frequently form and play a fundamental role in mitochondrial networks by restricting mitochondrial motility. Inter-mitochondrial contact untethering events are marked and regulated by mitochondria-lysosome contacts, which are modulated by RAB7 GTP hydrolysis. Moreover, inter-mitochondrial contact formation and untethering are further regulated by Mfn1/2 and Drp1 GTP hydrolysis, respectively. Surprisingly, endoplasmic reticulum tubules are also present at inter-mitochondrial contact untethering events, in addition to mitochondrial fission and fusion events. Importantly, we find that multiple Charcot-Marie-Tooth type 2 disease-linked mutations in Mfn2 (CMT2A), RAB7 (CMT2B), and TRPV4 (CMT2C) converge on prolonged inter-mitochondrial contacts and defective mitochondrial motility, highlighting a role for inter-mitochondrial contacts in mitochondrial network regulation and disease.

Indexed as

Mitochondrial DynamicsCharcot-Marie-Tooth DiseaseEndoplasmic ReticulumGTP PhosphohydrolasesHEK293 CellsHeLa CellsHumansLysosomesMitochondriaMitochondrial Proteinsrab7 GTP-Binding Proteinsrab GTP-Binding ProteinsTRPV Cation ChannelsGTP PhosphohydrolasesMFN2 protein, humanMitochondrial Proteinsrab7 GTP-Binding Proteinsrab7 GTP-binding proteins, humanrab GTP-Binding ProteinsTRPV4 protein, humanTRPV Cation ChannelsCharcot-Marie-Tooth type 2endoplasmic reticuluminter-mitochondrial contactlysosomeMfn2mitochondriamitochondria-lysosome contactRAB7super-resolution imagingTRPV4

Identifiers

PMID31231042
PMCPMC6726396
OpenAlexW2952155439

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.