The adhesion G protein-coupled receptor VLGR1/ADGRV1 controls autophagy

Publication type:
Journal article
Metadata:
Autoren
  • Joshua Linnert
  • Baran E Gueler
  • Jacek Krzysko
  • Uwe Wolfrum
Autoren-URL
https://www.webofscience.com/api/gateway?GWVersion=2&SrcApp=fis-test-1&SrcAuth=WosAPI&KeyUT=WOS:000971690000001&DestLinkType=FullRecord&DestApp=WOS_CPL
DOI
10.1111/bcpt.13869
eISSN
1742-7843
Externe Identifier
  • Clarivate Analytics Document Solution ID: U2AP0
  • PubMed Identifier: 37002809
ISSN
1742-7835
Ausgabe der Veröffentlichung
4
Zeitschrift
BASIC & CLINICAL PHARMACOLOGY & TOXICOLOGY
Schlüsselwörter
  • adhesion GPCR
  • affinity proteomics
  • autophagy
  • proteostasis
  • usher syndrome
Paginierung
313 - 330
Datum der Veröffentlichung
2023
Status
Published
Titel
The adhesion G protein-coupled receptor VLGR1/ADGRV1 controls autophagy
Sub types
  • Article
Ausgabe der Zeitschrift
133

Data source: Web of Science (Lite)

Other metadata sources:
Abstract
<jats:title>Abstract</jats:title><jats:p>VLGR1/ADGRV1 (very large G protein‐coupled receptor‐1) is the largest known adhesion G protein‐coupled receptor. Mutations in <jats:italic>VLGR1</jats:italic>/<jats:italic>ADGRV1</jats:italic> cause Usher syndrome (USH), the most common form of hereditary deaf‐blindness, and have been additionally linked to epilepsy. Although <jats:italic>VLGR1/ADGRV1</jats:italic> is almost ubiquitously expressed, little is known about the subcellular function and signalling of the VLGR1 protein and thus about mechanisms underlying the development of diseases. Using affinity proteomics, we identified key components of autophagosomes as putative interacting proteins of VLGR1. In addition, whole transcriptome sequencing of the retinae of the Vlgr1/del7TM mouse model revealed altered expression profiles of gene‐related autophagy. Monitoring autophagy by immunoblotting and immunocytochemistry of the LC3 and p62 as autophagy marker proteins revealed evoked autophagy in VLGR1‐deficient hTERT‐RPE1 cells and USH2C patient‐derived fibroblasts. Our data demonstrate the molecular and functional interaction of VLGR1 with key components of the autophagy process and point to an essential role of VLGR1 in the regulation of autophagy at internal membranes. The close association of VLGR1 with autophagy helps to explain the pathomechanisms underlying human USH and epilepsy related to VLGR1 defects.</jats:p>
Autoren
  • Joshua Linnert
  • Baran E Güler
  • Jacek Krzysko
  • Uwe Wolfrum
DOI
10.1111/bcpt.13869
eISSN
1742-7843
ISSN
1742-7835
Ausgabe der Veröffentlichung
4
Zeitschrift
Basic &amp; Clinical Pharmacology &amp; Toxicology
Sprache
en
Online publication date
2023
Paginierung
313 - 330
Datum der Veröffentlichung
2023
Status
Published
Herausgeber
Wiley
Herausgeber URL
http://dx.doi.org/10.1111/bcpt.13869
Datum der Datenerfassung
2023
Titel
The adhesion G protein‐coupled receptor VLGR1/ADGRV1 controls autophagy
Ausgabe der Zeitschrift
133

Data source: Crossref

Abstract
VLGR1/ADGRV1 (very large G protein-coupled receptor-1) is the largest known adhesion G protein-coupled receptor. Mutations in VLGR1/ADGRV1 cause Usher syndrome (USH), the most common form of hereditary deaf-blindness, and have been additionally linked to epilepsy. Although VLGR1/ADGRV1 is almost ubiquitously expressed, little is known about the subcellular function and signalling of the VLGR1 protein and thus about mechanisms underlying the development of diseases. Using affinity proteomics, we identified key components of autophagosomes as putative interacting proteins of VLGR1. In addition, whole transcriptome sequencing of the retinae of the Vlgr1/del7TM mouse model revealed altered expression profiles of gene-related autophagy. Monitoring autophagy by immunoblotting and immunocytochemistry of the LC3 and p62 as autophagy marker proteins revealed evoked autophagy in VLGR1-deficient hTERT-RPE1 cells and USH2C patient-derived fibroblasts. Our data demonstrate the molecular and functional interaction of VLGR1 with key components of the autophagy process and point to an essential role of VLGR1 in the regulation of autophagy at internal membranes. The close association of VLGR1 with autophagy helps to explain the pathomechanisms underlying human USH and epilepsy related to VLGR1 defects.
Addresses
  • Institute of Molecular Physiology, Molecular Cell Biology, Johannes Gutenberg University Mainz, Mainz, Germany.
Autoren
  • Joshua Linnert
  • Baran E Güler
  • Jacek Krzysko
  • Uwe Wolfrum
DOI
10.1111/bcpt.13869
eISSN
1742-7843
Externe Identifier
  • PubMed Identifier: 37002809
Funding acknowledgements
  • Foundation Fighting Blindness: PPA‐0717‐0719‐RAD
  • inneruniversitäre Forschungsförderung ("Stufe I") of the Johannes Gutenberg University Mainz (UW):
  • Foundation Fighting Blindness (FFB): (UW)
  • German Research Council DFG FOR 2149 Elucidation of Adhesion-GPCR Signaling: 246212759
  • Foundation Fighting Blindness (FFB): PPA-0717-0719-RAD
  • German Research Council DFG FOR 2149 Elucidation of Adhesion-GPCR Signaling: (UW)
Open access
false
ISSN
1742-7835
Ausgabe der Veröffentlichung
4
Zeitschrift
Basic & clinical pharmacology & toxicology
Schlüsselwörter
  • Animals
  • Humans
  • Mice
  • Epilepsy
  • Receptors, G-Protein-Coupled
  • Signal Transduction
  • Autophagy
  • Usher Syndromes
Sprache
eng
Medium
Print-Electronic
Online publication date
2023
Paginierung
313 - 330
Datum der Veröffentlichung
2023
Status
Published
Publisher licence
CC BY-NC
Datum der Datenerfassung
2023
Titel
The adhesion G protein-coupled receptor VLGR1/ADGRV1 controls autophagy.
Sub types
  • Journal Article
Ausgabe der Zeitschrift
133

Data source: Europe PubMed Central

Abstract
VLGR1/ADGRV1 (very large G protein-coupled receptor-1) is the largest known adhesion G protein-coupled receptor. Mutations in VLGR1/ADGRV1 cause Usher syndrome (USH), the most common form of hereditary deaf-blindness, and have been additionally linked to epilepsy. Although VLGR1/ADGRV1 is almost ubiquitously expressed, little is known about the subcellular function and signalling of the VLGR1 protein and thus about mechanisms underlying the development of diseases. Using affinity proteomics, we identified key components of autophagosomes as putative interacting proteins of VLGR1. In addition, whole transcriptome sequencing of the retinae of the Vlgr1/del7TM mouse model revealed altered expression profiles of gene-related autophagy. Monitoring autophagy by immunoblotting and immunocytochemistry of the LC3 and p62 as autophagy marker proteins revealed evoked autophagy in VLGR1-deficient hTERT-RPE1 cells and USH2C patient-derived fibroblasts. Our data demonstrate the molecular and functional interaction of VLGR1 with key components of the autophagy process and point to an essential role of VLGR1 in the regulation of autophagy at internal membranes. The close association of VLGR1 with autophagy helps to explain the pathomechanisms underlying human USH and epilepsy related to VLGR1 defects.
Date of acceptance
2023
Autoren
  • Joshua Linnert
  • Baran E Güler
  • Jacek Krzysko
  • Uwe Wolfrum
Autoren-URL
https://www.ncbi.nlm.nih.gov/pubmed/37002809
DOI
10.1111/bcpt.13869
eISSN
1742-7843
Funding acknowledgements
  • German Research Council DFG FOR 2149 Elucidation of Adhesion-GPCR Signaling: 246212759
  • German Research Council DFG FOR 2149 Elucidation of Adhesion-GPCR Signaling: (UW)
  • Foundation Fighting Blindness (FFB): PPA-0717-0719-RAD
  • Foundation Fighting Blindness (FFB): (UW)
  • inneruniversitäre Forschungsförderung ("Stufe I") of the Johannes Gutenberg University Mainz (UW):
Ausgabe der Veröffentlichung
4
Zeitschrift
Basic Clin Pharmacol Toxicol
Schlüsselwörter
  • adhesion GPCR
  • affinity proteomics
  • autophagy
  • proteostasis
  • usher syndrome
  • Animals
  • Humans
  • Mice
  • Autophagy
  • Epilepsy
  • Receptors, G-Protein-Coupled
  • Signal Transduction
  • Usher Syndromes
Sprache
eng
Country
England
Paginierung
313 - 330
Datum der Veröffentlichung
2023
Status
Published
Datum, an dem der Datensatz öffentlich gemacht wurde
2023
Titel
The adhesion G protein-coupled receptor VLGR1/ADGRV1 controls autophagy.
Sub types
  • Journal Article
Ausgabe der Zeitschrift
133

Data source: PubMed

Author's licence
CC-BY-NC
Autoren
  • Joshua Linnert
  • Baran E Güler
  • Jacek Krzysko
  • Uwe Wolfrum
Hosting institution
Universitätsbibliothek Mainz
Sammlungen
  • DFG-491381577-H
Resource version
Published version
DOI
10.1111/bcpt.13869
File(s) embargoed
false
Open access
true
ISSN
1742-7835
Zeitschrift
Basic & clinical pharmacology & toxicology
Schlüsselwörter
  • 570 Biowissenschaften
  • 570 Life sciences
Sprache
eng
Open access status
Open Access
Datum der Veröffentlichung
2023
Public URL
https://openscience.ub.uni-mainz.de/handle/20.500.12030/9243
Herausgeber
Wiley-Blackwell
Datum der Datenerfassung
2023
Datum, an dem der Datensatz öffentlich gemacht wurde
2023
Zugang
Public
Titel
The adhesion G protein-coupled receptor VLGR1/ADGRV1 controls autophagy
Ausgabe der Zeitschrift
Version of Record (VoR)

Files

the_adhesion_g_proteincoupled-20230607095356843.pdf

Data source: OPENSCIENCE.UB

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