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GIMAP6 regulates autophagy, immune competence, and inflammation in mice and humans
Inborn errors of immunity (IEIs) unveil regulatory pathways of human immunity. We describe a new IEI caused by mutations in the GTPase of the immune-associated protein 6 (GIMAP6) gene in patients with infections, lymphoproliferation, autoimmunity, and multiorgan vasculitis. Patients and Gimap6(−/−)...
| Autores principales: | , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , |
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| Formato: | Online Artículo Texto |
| Lenguaje: | English |
| Publicado: |
Rockefeller University Press
2022
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| Materias: | |
| Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9111091/ https://www.ncbi.nlm.nih.gov/pubmed/35551368 http://dx.doi.org/10.1084/jem.20201405 |
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| author | Yao, Yikun Du Jiang, Ping Chao, Brittany N. Cagdas, Deniz Kubo, Satoshi Balasubramaniyam, Arasu Zhang, Yu Shadur, Bella NaserEddin, Adeeb Folio, Les R. Schwarz, Benjamin Bohrnsen, Eric Zheng, Lixin Lynberg, Matthew Gottlieb, Simone Leney-Greene, Michael A. Park, Ann Y. Tezcan, Ilhan Akdogan, Ali Gocmen, Rahsan Onder, Sevgen Rosenberg, Avi Soilleux, Elizabeth J. Johnson, Errin Jackson, Peter K. Demeter, Janos Chauvin, Samuel D. Paul, Florian Selbach, Matthias Bulut, Haydar Clatworthy, Menna R. Tuong, Zewen K. Zhang, Hanlin Stewart, Benjamin J. Bosio, Catharine M. Stepensky, Polina Clare, Simon Ganesan, Sundar Pascall, John C. Daumke, Oliver Butcher, Geoffrey W. McMichael, Andrew J. Simon, Anna Katharina Lenardo, Michael J. |
| author_facet | Yao, Yikun Du Jiang, Ping Chao, Brittany N. Cagdas, Deniz Kubo, Satoshi Balasubramaniyam, Arasu Zhang, Yu Shadur, Bella NaserEddin, Adeeb Folio, Les R. Schwarz, Benjamin Bohrnsen, Eric Zheng, Lixin Lynberg, Matthew Gottlieb, Simone Leney-Greene, Michael A. Park, Ann Y. Tezcan, Ilhan Akdogan, Ali Gocmen, Rahsan Onder, Sevgen Rosenberg, Avi Soilleux, Elizabeth J. Johnson, Errin Jackson, Peter K. Demeter, Janos Chauvin, Samuel D. Paul, Florian Selbach, Matthias Bulut, Haydar Clatworthy, Menna R. Tuong, Zewen K. Zhang, Hanlin Stewart, Benjamin J. Bosio, Catharine M. Stepensky, Polina Clare, Simon Ganesan, Sundar Pascall, John C. Daumke, Oliver Butcher, Geoffrey W. McMichael, Andrew J. Simon, Anna Katharina Lenardo, Michael J. |
| author_sort | Yao, Yikun |
| collection | PubMed |
| description | Inborn errors of immunity (IEIs) unveil regulatory pathways of human immunity. We describe a new IEI caused by mutations in the GTPase of the immune-associated protein 6 (GIMAP6) gene in patients with infections, lymphoproliferation, autoimmunity, and multiorgan vasculitis. Patients and Gimap6(−/−) mice show defects in autophagy, redox regulation, and polyunsaturated fatty acid (PUFA)–containing lipids. We find that GIMAP6 complexes with GABARAPL2 and GIMAP7 to regulate GTPase activity. Also, GIMAP6 is induced by IFN-γ and plays a critical role in antibacterial immunity. Finally, we observed that Gimap6(−/−) mice died prematurely from microangiopathic glomerulosclerosis most likely due to GIMAP6 deficiency in kidney endothelial cells. |
| format | Online Article Text |
| id | pubmed-9111091 |
| institution | National Center for Biotechnology Information |
| language | English |
| publishDate | 2022 |
| publisher | Rockefeller University Press |
| record_format | MEDLINE/PubMed |
| spelling | pubmed-91110912022-11-15 GIMAP6 regulates autophagy, immune competence, and inflammation in mice and humans Yao, Yikun Du Jiang, Ping Chao, Brittany N. Cagdas, Deniz Kubo, Satoshi Balasubramaniyam, Arasu Zhang, Yu Shadur, Bella NaserEddin, Adeeb Folio, Les R. Schwarz, Benjamin Bohrnsen, Eric Zheng, Lixin Lynberg, Matthew Gottlieb, Simone Leney-Greene, Michael A. Park, Ann Y. Tezcan, Ilhan Akdogan, Ali Gocmen, Rahsan Onder, Sevgen Rosenberg, Avi Soilleux, Elizabeth J. Johnson, Errin Jackson, Peter K. Demeter, Janos Chauvin, Samuel D. Paul, Florian Selbach, Matthias Bulut, Haydar Clatworthy, Menna R. Tuong, Zewen K. Zhang, Hanlin Stewart, Benjamin J. Bosio, Catharine M. Stepensky, Polina Clare, Simon Ganesan, Sundar Pascall, John C. Daumke, Oliver Butcher, Geoffrey W. McMichael, Andrew J. Simon, Anna Katharina Lenardo, Michael J. J Exp Med Article Inborn errors of immunity (IEIs) unveil regulatory pathways of human immunity. We describe a new IEI caused by mutations in the GTPase of the immune-associated protein 6 (GIMAP6) gene in patients with infections, lymphoproliferation, autoimmunity, and multiorgan vasculitis. Patients and Gimap6(−/−) mice show defects in autophagy, redox regulation, and polyunsaturated fatty acid (PUFA)–containing lipids. We find that GIMAP6 complexes with GABARAPL2 and GIMAP7 to regulate GTPase activity. Also, GIMAP6 is induced by IFN-γ and plays a critical role in antibacterial immunity. Finally, we observed that Gimap6(−/−) mice died prematurely from microangiopathic glomerulosclerosis most likely due to GIMAP6 deficiency in kidney endothelial cells. Rockefeller University Press 2022-05-13 /pmc/articles/PMC9111091/ /pubmed/35551368 http://dx.doi.org/10.1084/jem.20201405 Text en This is a work of the U.S. Government and is not subject to copyright protection in the United States. Foreign copyrights may apply. https://creativecommons.org/licenses/by-nc-sa/4.0/http://www.rupress.org/terms/This article is distributed under the terms of an Attribution–Noncommercial–Share Alike–No Mirror Sites license for the first six months after the publication date (see http://www.rupress.org/terms/). After six months it is available under a Creative Commons License (Attribution–Noncommercial–Share Alike 4.0 International license, as described at https://creativecommons.org/licenses/by-nc-sa/4.0/). |
| spellingShingle | Article Yao, Yikun Du Jiang, Ping Chao, Brittany N. Cagdas, Deniz Kubo, Satoshi Balasubramaniyam, Arasu Zhang, Yu Shadur, Bella NaserEddin, Adeeb Folio, Les R. Schwarz, Benjamin Bohrnsen, Eric Zheng, Lixin Lynberg, Matthew Gottlieb, Simone Leney-Greene, Michael A. Park, Ann Y. Tezcan, Ilhan Akdogan, Ali Gocmen, Rahsan Onder, Sevgen Rosenberg, Avi Soilleux, Elizabeth J. Johnson, Errin Jackson, Peter K. Demeter, Janos Chauvin, Samuel D. Paul, Florian Selbach, Matthias Bulut, Haydar Clatworthy, Menna R. Tuong, Zewen K. Zhang, Hanlin Stewart, Benjamin J. Bosio, Catharine M. Stepensky, Polina Clare, Simon Ganesan, Sundar Pascall, John C. Daumke, Oliver Butcher, Geoffrey W. McMichael, Andrew J. Simon, Anna Katharina Lenardo, Michael J. GIMAP6 regulates autophagy, immune competence, and inflammation in mice and humans |
| title | GIMAP6 regulates autophagy, immune competence, and inflammation in mice and humans |
| title_full | GIMAP6 regulates autophagy, immune competence, and inflammation in mice and humans |
| title_fullStr | GIMAP6 regulates autophagy, immune competence, and inflammation in mice and humans |
| title_full_unstemmed | GIMAP6 regulates autophagy, immune competence, and inflammation in mice and humans |
| title_short | GIMAP6 regulates autophagy, immune competence, and inflammation in mice and humans |
| title_sort | gimap6 regulates autophagy, immune competence, and inflammation in mice and humans |
| topic | Article |
| url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9111091/ https://www.ncbi.nlm.nih.gov/pubmed/35551368 http://dx.doi.org/10.1084/jem.20201405 |
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