Cargando…
Mutation in VPS35 associated with Parkinson’s disease impairs WASH complex association and inhibits autophagy
Endosomal protein sorting controls the localization of many physiologically important proteins and is linked to several neurodegenerative diseases. VPS35 is a component of the retromer complex, which mediates endosome-to-Golgi retrieval of membrane proteins such as the cation-independent mannose 6-p...
Autores principales: | , , , , , , |
---|---|
Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Nature Pub. Group
2014
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4024763/ https://www.ncbi.nlm.nih.gov/pubmed/24819384 http://dx.doi.org/10.1038/ncomms4828 |
_version_ | 1782316691851575296 |
---|---|
author | Zavodszky, Eszter Seaman, Matthew N.J. Moreau, Kevin Jimenez-Sanchez, Maria Breusegem, Sophia Y. Harbour, Michael E. Rubinsztein, David C. |
author_facet | Zavodszky, Eszter Seaman, Matthew N.J. Moreau, Kevin Jimenez-Sanchez, Maria Breusegem, Sophia Y. Harbour, Michael E. Rubinsztein, David C. |
author_sort | Zavodszky, Eszter |
collection | PubMed |
description | Endosomal protein sorting controls the localization of many physiologically important proteins and is linked to several neurodegenerative diseases. VPS35 is a component of the retromer complex, which mediates endosome-to-Golgi retrieval of membrane proteins such as the cation-independent mannose 6-phosphate receptor. Furthermore, retromer is also required for the endosomal recruitment of the actin nucleation promoting WASH complex. The VPS35 D620N mutation causes a rare form of autosomal-dominant Parkinson’s disease (PD). Here we show that this mutant associates poorly with the WASH complex and impairs WASH recruitment to endosomes. Autophagy is impaired in cells expressing PD-mutant VPS35 or lacking WASH. The autophagy defects can be explained, at least in part, by abnormal trafficking of the autophagy protein ATG9A. Thus, the PD-causing D620N mutation in VPS35 restricts WASH complex recruitment to endosomes, and reveals a novel role for the WASH complex in autophagosome formation. |
format | Online Article Text |
id | pubmed-4024763 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2014 |
publisher | Nature Pub. Group |
record_format | MEDLINE/PubMed |
spelling | pubmed-40247632014-05-20 Mutation in VPS35 associated with Parkinson’s disease impairs WASH complex association and inhibits autophagy Zavodszky, Eszter Seaman, Matthew N.J. Moreau, Kevin Jimenez-Sanchez, Maria Breusegem, Sophia Y. Harbour, Michael E. Rubinsztein, David C. Nat Commun Article Endosomal protein sorting controls the localization of many physiologically important proteins and is linked to several neurodegenerative diseases. VPS35 is a component of the retromer complex, which mediates endosome-to-Golgi retrieval of membrane proteins such as the cation-independent mannose 6-phosphate receptor. Furthermore, retromer is also required for the endosomal recruitment of the actin nucleation promoting WASH complex. The VPS35 D620N mutation causes a rare form of autosomal-dominant Parkinson’s disease (PD). Here we show that this mutant associates poorly with the WASH complex and impairs WASH recruitment to endosomes. Autophagy is impaired in cells expressing PD-mutant VPS35 or lacking WASH. The autophagy defects can be explained, at least in part, by abnormal trafficking of the autophagy protein ATG9A. Thus, the PD-causing D620N mutation in VPS35 restricts WASH complex recruitment to endosomes, and reveals a novel role for the WASH complex in autophagosome formation. Nature Pub. Group 2014-05-13 /pmc/articles/PMC4024763/ /pubmed/24819384 http://dx.doi.org/10.1038/ncomms4828 Text en Copyright © 2014, Nature Publishing Group, a division of Macmillan Publishers Limited. All Rights Reserved. http://creativecommons.org/licenses/by/3.0/ This work is licensed under a Creative Commons Attribution 3.0 Unported License. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in the credit line; if the material is not included under the Creative Commons license, users will need to obtain permission from the license holder to reproduce the material. To view a copy of this license, visit http://creativecommons.org/licenses/by/3.0/ |
spellingShingle | Article Zavodszky, Eszter Seaman, Matthew N.J. Moreau, Kevin Jimenez-Sanchez, Maria Breusegem, Sophia Y. Harbour, Michael E. Rubinsztein, David C. Mutation in VPS35 associated with Parkinson’s disease impairs WASH complex association and inhibits autophagy |
title | Mutation in VPS35 associated with Parkinson’s disease impairs WASH complex association and inhibits autophagy |
title_full | Mutation in VPS35 associated with Parkinson’s disease impairs WASH complex association and inhibits autophagy |
title_fullStr | Mutation in VPS35 associated with Parkinson’s disease impairs WASH complex association and inhibits autophagy |
title_full_unstemmed | Mutation in VPS35 associated with Parkinson’s disease impairs WASH complex association and inhibits autophagy |
title_short | Mutation in VPS35 associated with Parkinson’s disease impairs WASH complex association and inhibits autophagy |
title_sort | mutation in vps35 associated with parkinson’s disease impairs wash complex association and inhibits autophagy |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4024763/ https://www.ncbi.nlm.nih.gov/pubmed/24819384 http://dx.doi.org/10.1038/ncomms4828 |
work_keys_str_mv | AT zavodszkyeszter mutationinvps35associatedwithparkinsonsdiseaseimpairswashcomplexassociationandinhibitsautophagy AT seamanmatthewnj mutationinvps35associatedwithparkinsonsdiseaseimpairswashcomplexassociationandinhibitsautophagy AT moreaukevin mutationinvps35associatedwithparkinsonsdiseaseimpairswashcomplexassociationandinhibitsautophagy AT jimenezsanchezmaria mutationinvps35associatedwithparkinsonsdiseaseimpairswashcomplexassociationandinhibitsautophagy AT breusegemsophiay mutationinvps35associatedwithparkinsonsdiseaseimpairswashcomplexassociationandinhibitsautophagy AT harbourmichaele mutationinvps35associatedwithparkinsonsdiseaseimpairswashcomplexassociationandinhibitsautophagy AT rubinszteindavidc mutationinvps35associatedwithparkinsonsdiseaseimpairswashcomplexassociationandinhibitsautophagy |