Evidence map›Paper›PMID 30044992›Full record

ArticleCell reports2018

The ESCRT-III Protein CHMP1A Mediates Secretion of Sonic Hedgehog on a Distinctive Subtype of Extracellular Vesicles.

Michael E Coulter, Cristina M Dorobantu, Gerrald A Lodewijk, François Delalande, Sarah Cianferani, Vijay S Ganesh, Richard S Smith, Elaine T Lim, C Shan Xu, Song Pang and 15 more

Open access · goldAbstract read
In one paragraph

Article in Cell reports, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 69 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
69citing papers in PubMed, 2 pooled it
4.7field-weighted citation impact, top 4% 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

69 citing papers in PubMed, 2 syntheses or guidelines pooled it, 104 citations in OpenAlex.

  1. Pooled it
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  11. Apocrine secretion by the choroid plexus.Fluids and barriers of the CNS · 2025
    Review
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  14. Astrocyte Modulation of Synaptic Plasticity Mediated by Activity-Dependent Sonic Hedgehog Signaling.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2025
    Article
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9 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

25 authors at 8 institutions in 3 countries.

Michael E CoulterDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Departments of Pediatrics and Neurology, Harvard Medical School, Boston, MA 02115, USA; Program in Neuroscience and Harvard/MIT MD-PHD Program, Harvard Medical School, Boston, MA 02115, USA.
Cristina M DorobantuInserm U1110, Université de Strasbourg, Institut de Recherche sur les Maladies Virales et Hépatiques, 67000 Strasbourg, France.
Gerrald A LodewijkUniversity of Amsterdam, Swammerdam Institute for Life Sciences, 1098 XH Amsterdam, the Netherlands.
François DelalandeLaboratoire de Spectrométrie de Masse Bio-Organique, IPHC, UMR 7178, CNRS-Université de Strasbourg, ECPM, 67087 Strasbourg, France.
Sarah CianferaniLaboratoire de Spectrométrie de Masse Bio-Organique, IPHC, UMR 7178, CNRS-Université de Strasbourg, ECPM, 67087 Strasbourg, France.
Vijay S GaneshDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Departments of Pediatrics and Neurology, Harvard Medical School, Boston, MA 02115, USA; Department of Neurology, Massachusetts General Hospital, Boston, MA 02114, USA; Department of Neurology, Brigham and Women's Hospital, Boston, MA 02115, USA.
Richard S SmithDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Departments of Pediatrics and Neurology, Harvard Medical School, Boston, MA 02115, USA.
Elaine T LimDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Departments of Pediatrics and Neurology, Harvard Medical School, Boston, MA 02115, USA.
C Shan XuJanelia Research Campus, Howard Hughes Medical Institute, Ashburn, VA 20147, USA.
Song PangJanelia Research Campus, Howard Hughes Medical Institute, Ashburn, VA 20147, USA.
Eric T WongBrain Tumor Center and Neuro-Oncology Unit, Beth Israel Deaconess Medical Center, Boston, MA 02215, USA.
Hart G W LidovDepartment of Pathology, Boston Children's Hospital, Boston, MA 02115, USA.
Monica L CalicchioDepartment of Pathology, Boston Children's Hospital, Boston, MA 02115, USA.
Edward YangDepartment of Radiology, Boston Children's Hospital, Boston, MA 02115, USA.
Dilenny M GonzalezDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Departments of Pediatrics and Neurology, Harvard Medical School, Boston, MA 02115, USA.
Thorsten M SchlaegerDivision of Hematology and Oncology, Boston Children's Hospital, Boston, MA 02115, USA.
Ganeshwaran H MochidaDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Departments of Pediatrics and Neurology, Harvard Medical School, Boston, MA 02115, USA; Department of Neurology, Massachusetts General Hospital, Boston, MA 02114, USA.
Harald HessJanelia Research Campus, Howard Hughes Medical Institute, Ashburn, VA 20147, USA.
Wei-Chung Allen LeeF.M. Kirby Neurobiology Center, Boston Children's Hospital and Department of Neurology, Harvard Medical School, Boston, MA 02115, USA.
Maria K LehtinenDepartment of Pathology, Boston Children's Hospital, Boston, MA 02115, USA.
Tomas KirchhausenProgram in Cellular and Molecular Medicine, Boston Children's Hospital and Department of Cell Biology, Harvard Medical School, Boston, MA 02115, USA; Department of Pediatrics, Harvard Medical School, Boston, MA 02115, USA.
David HausslerCenter for Biomolecular Science and Engineering, University of California and Howard Hughes Medical Institute, Santa Cruz, CA 95064, USA.
Frank M J JacobsUniversity of Amsterdam, Swammerdam Institute for Life Sciences, 1098 XH Amsterdam, the Netherlands. Electronic address: f.m.j.jacobs@uva.nl.
Raphael GaudinInserm U1110, Université de Strasbourg, Institut de Recherche sur les Maladies Virales et Hépatiques, 67000 Strasbourg, France; Program in Cellular and Molecular Medicine, Boston Children's Hospital and Department of Cell Biology, Harvard Medical School, Boston, MA 02115, USA. Electronic address: rgaudin@unistra.fr.
Christopher A WalshDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Departments of Pediatrics and Neurology, Harvard Medical School, Boston, MA 02115, USA. Electronic address: christopher.walsh@childrens.harvard.edu.
Boston Children's Hospital · USHarvard University · USHoward Hughes Medical Institute · USJanelia Research Campus · USInstitute for Translational Medicine and Liver DiseaseUniversité de Strasbourg · FRUniversity of Amsterdam · NLBeth Israel Deaconess Medical Center · US

Funding

Small-molecule inhibitors of flavivirus entryU19AI109740 · NIAID · HARVARD MEDICAL SCHOOL · PI WHELAN, SEAN PJ · 2014 to 2018
$23.0M
HUMAN EPILEPSY GENETICS--NEURONAL MIGRATION DISORDERSR01NS035129 · NINDS · BOSTON CHILDREN'S HOSPITAL · PI WALSH, CHRISTOPHER A. · 1997 to 2023
$9.5M
Control of neural stem cells by the nascent cerebrospinal fluidR01NS088566 · NINDS · BOSTON CHILDREN'S HOSPITAL · PI LEHTINEN, MARIA · 2014 to 2024
$5.5M
Mouse Neurodevelopmental Behavior CoreU54HD090255 · NICHD · BOSTON CHILDREN'S HOSPITAL · PI SAHIN, MUSTAFA · 2016 to 2020
$5.2M
Dynamics of Clathrin Coat Formation in CellsR01GM075252 · NIGMS · IMMUNE DISEASE INSTITUTE, INC. · PI KIRCHHAUSEN, TOMAS · 2005 to 2018
$4.6M
Role for ion conducting proteins in cortical malformation diseasesK99NS112604 · NINDS · BOSTON CHILDREN'S HOSPITAL · PI SMITH, RICHARD S · 2019 to 2021
$276k
A Novel Essential Gene for Human Cognitive FunctionF30MH102909 · NIMH · HARVARD MEDICAL SCHOOL · PI COULTER, MICHAEL EDWARD · 2013 to 2017
$173k
Cerebral Cortex Malformations - Genetics and Physiological ValidationF32NS100338 · NINDS · BOSTON CHILDREN'S HOSPITAL · PI SMITH, RICHARD S · 2017 to 2019
$161k
Howard Hughes Medical InstituteNIAID NIH HHS U19 AI109740NICHD NIH HHS U54 HD090255NIGMS NIH HHS R01 GM075252NIMH NIH HHS F30 MH102909NINDS NIH HHS F32 NS100338NINDS NIH HHS K99 NS112604NINDS NIH HHS R01 NS035129NINDS NIH HHS R01 NS088566
6 · The paper itself

Abstract

Endosomal sorting complex required for transport (ESCRT) complex proteins regulate biogenesis and release of extracellular vesicles (EVs), which enable cell-to-cell communication in the nervous system essential for development and adult function. We recently showed human loss-of-function (LOF) mutations in ESCRT-III member CHMP1A cause autosomal recessive microcephaly with pontocerebellar hypoplasia, but its mechanism was unclear. Here, we show Chmp1a is required for progenitor proliferation in mouse cortex and cerebellum and progenitor maintenance in human cerebral organoids. In Chmp1a null mice, this defect is associated with impaired sonic hedgehog (Shh) secretion and intraluminal vesicle (ILV) formation in multivesicular bodies (MVBs). Furthermore, we show CHMP1A is important for release of an EV subtype that contains AXL, RAB18, and TMED10 (ART) and SHH. Our findings show CHMP1A loss impairs secretion of SHH on ART-EVs, providing molecular mechanistic insights into the role of ESCRT proteins and EVs in the brain.

Indexed as

AdultAnimalsBrainChoroid PlexusEndosomal Sorting Complexes Required for TransportExtracellular VesiclesHedgehog ProteinsHumansInfant, NewbornMiceNIH 3T3 CellsVesicular Transport ProteinsCHMP1A protein, humanEndosomal Sorting Complexes Required for TransportHedgehog ProteinsSHH protein, humanVesicular Transport ProteinsCHMP1AESCRTextracellular vesiclesmicrocephalymultivesicular bodyneurodevelopmentsonic hedgehog

Identifiers

PMID30044992
PMCPMC6178983
OpenAlexW2884643428

What OpenQuestion holds

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LicenceCC BY
Read underepoch 390

Registered trials

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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.