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Distinct partitioning of ALS associated TDP-43, FUS and SOD1 mutants into cellular inclusions
Amyotrophic lateral sclerosis is a rapidly progressing neurodegenerative disease associated with protein misfolding and aggregation. Most cases are characterized by TDP-43 positive inclusions, while a minority of familial ALS cases are instead FUS and SOD1 positive respectively. Cells can generate i...
Autores principales: | , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Nature Publishing Group
2015
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4544019/ https://www.ncbi.nlm.nih.gov/pubmed/26293199 http://dx.doi.org/10.1038/srep13416 |
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author | Farrawell, Natalie E. Lambert-Smith, Isabella A. Warraich, Sadaf T. Blair, Ian P. Saunders, Darren N. Hatters, Danny M. Yerbury, Justin J. |
author_facet | Farrawell, Natalie E. Lambert-Smith, Isabella A. Warraich, Sadaf T. Blair, Ian P. Saunders, Darren N. Hatters, Danny M. Yerbury, Justin J. |
author_sort | Farrawell, Natalie E. |
collection | PubMed |
description | Amyotrophic lateral sclerosis is a rapidly progressing neurodegenerative disease associated with protein misfolding and aggregation. Most cases are characterized by TDP-43 positive inclusions, while a minority of familial ALS cases are instead FUS and SOD1 positive respectively. Cells can generate inclusions of variable type including previously characterized aggresomes, IPOD or JUNQ structures depending on the misfolded protein. SOD1 invariably forms JUNQ inclusions but it remains unclear whether other ALS protein aggregates arise as one of these previously described inclusion types or form unique structures. Here we show that FUS variably partitioned to IPOD, JUNQ or alternate structures, contain a mobile fraction, were not microtubule dependent and initially did not contain ubiquitin. TDP-43 inclusions formed in a microtubule independent manner, did not contain a mobile fraction but variably colocalized to JUNQ inclusions and another alternate structure. We conclude that the RNA binding proteins TDP-43 and FUS do not consistently fit the currently characterised inclusion models suggesting that cells have a larger repertoire for generating inclusions than currently thought, and imply that toxicity in ALS does not stem from a particular aggregation process or aggregate structure. |
format | Online Article Text |
id | pubmed-4544019 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2015 |
publisher | Nature Publishing Group |
record_format | MEDLINE/PubMed |
spelling | pubmed-45440192015-09-01 Distinct partitioning of ALS associated TDP-43, FUS and SOD1 mutants into cellular inclusions Farrawell, Natalie E. Lambert-Smith, Isabella A. Warraich, Sadaf T. Blair, Ian P. Saunders, Darren N. Hatters, Danny M. Yerbury, Justin J. Sci Rep Article Amyotrophic lateral sclerosis is a rapidly progressing neurodegenerative disease associated with protein misfolding and aggregation. Most cases are characterized by TDP-43 positive inclusions, while a minority of familial ALS cases are instead FUS and SOD1 positive respectively. Cells can generate inclusions of variable type including previously characterized aggresomes, IPOD or JUNQ structures depending on the misfolded protein. SOD1 invariably forms JUNQ inclusions but it remains unclear whether other ALS protein aggregates arise as one of these previously described inclusion types or form unique structures. Here we show that FUS variably partitioned to IPOD, JUNQ or alternate structures, contain a mobile fraction, were not microtubule dependent and initially did not contain ubiquitin. TDP-43 inclusions formed in a microtubule independent manner, did not contain a mobile fraction but variably colocalized to JUNQ inclusions and another alternate structure. We conclude that the RNA binding proteins TDP-43 and FUS do not consistently fit the currently characterised inclusion models suggesting that cells have a larger repertoire for generating inclusions than currently thought, and imply that toxicity in ALS does not stem from a particular aggregation process or aggregate structure. Nature Publishing Group 2015-08-21 /pmc/articles/PMC4544019/ /pubmed/26293199 http://dx.doi.org/10.1038/srep13416 Text en Copyright © 2015, Macmillan Publishers Limited http://creativecommons.org/licenses/by/4.0/ This work is licensed under a Creative Commons Attribution 4.0 International 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/4.0/ |
spellingShingle | Article Farrawell, Natalie E. Lambert-Smith, Isabella A. Warraich, Sadaf T. Blair, Ian P. Saunders, Darren N. Hatters, Danny M. Yerbury, Justin J. Distinct partitioning of ALS associated TDP-43, FUS and SOD1 mutants into cellular inclusions |
title | Distinct partitioning of ALS associated TDP-43, FUS and SOD1 mutants into cellular inclusions |
title_full | Distinct partitioning of ALS associated TDP-43, FUS and SOD1 mutants into cellular inclusions |
title_fullStr | Distinct partitioning of ALS associated TDP-43, FUS and SOD1 mutants into cellular inclusions |
title_full_unstemmed | Distinct partitioning of ALS associated TDP-43, FUS and SOD1 mutants into cellular inclusions |
title_short | Distinct partitioning of ALS associated TDP-43, FUS and SOD1 mutants into cellular inclusions |
title_sort | distinct partitioning of als associated tdp-43, fus and sod1 mutants into cellular inclusions |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4544019/ https://www.ncbi.nlm.nih.gov/pubmed/26293199 http://dx.doi.org/10.1038/srep13416 |
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