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Genetic inactivation of RIP1 kinase activity in rats protects against ischemic brain injury

RIP1 kinase-mediated inflammatory and cell death pathways have been implicated in the pathology of acute and chronic disorders of the nervous system. Here, we describe a novel animal model of RIP1 kinase deficiency, generated by knock-in of the kinase-inactivating RIP1(D138N) mutation in rats. Homoz...

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Autores principales: Stark, Kimberly, Goncharov, Tatiana, Varfolomeev, Eugene, Xie, Luke, Ngu, Hai, Peng, Ivan, Anderson, Keith R., Verschueren, Erik, Choi, Meena, Kirkpatrick, Donald S., Easton, Amy, Webster, Joshua D., McKenzie, Brent S., Vucic, Domagoj, Bingol, Baris
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group UK 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8026634/
https://www.ncbi.nlm.nih.gov/pubmed/33828080
http://dx.doi.org/10.1038/s41419-021-03651-6
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author Stark, Kimberly
Goncharov, Tatiana
Varfolomeev, Eugene
Xie, Luke
Ngu, Hai
Peng, Ivan
Anderson, Keith R.
Verschueren, Erik
Choi, Meena
Kirkpatrick, Donald S.
Easton, Amy
Webster, Joshua D.
McKenzie, Brent S.
Vucic, Domagoj
Bingol, Baris
author_facet Stark, Kimberly
Goncharov, Tatiana
Varfolomeev, Eugene
Xie, Luke
Ngu, Hai
Peng, Ivan
Anderson, Keith R.
Verschueren, Erik
Choi, Meena
Kirkpatrick, Donald S.
Easton, Amy
Webster, Joshua D.
McKenzie, Brent S.
Vucic, Domagoj
Bingol, Baris
author_sort Stark, Kimberly
collection PubMed
description RIP1 kinase-mediated inflammatory and cell death pathways have been implicated in the pathology of acute and chronic disorders of the nervous system. Here, we describe a novel animal model of RIP1 kinase deficiency, generated by knock-in of the kinase-inactivating RIP1(D138N) mutation in rats. Homozygous RIP1 kinase-dead (KD) rats had normal development, reproduction and did not show any gross phenotypes at baseline. However, cells derived from RIP1 KD rats displayed resistance to necroptotic cell death. In addition, RIP1 KD rats were resistant to TNF-induced systemic shock. We studied the utility of RIP1 KD rats for neurological disorders by testing the efficacy of the genetic inactivation in the transient middle cerebral artery occlusion/reperfusion model of brain injury. RIP1 KD rats were protected in this model in a battery of behavioral, imaging, and histopathological endpoints. In addition, RIP1 KD rats had reduced inflammation and accumulation of neuronal injury biomarkers. Unbiased proteomics in the plasma identified additional changes that were ameliorated by RIP1 genetic inactivation. Together these data highlight the utility of the RIP1 KD rats for target validation and biomarker studies for neurological disorders.
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spelling pubmed-80266342021-04-21 Genetic inactivation of RIP1 kinase activity in rats protects against ischemic brain injury Stark, Kimberly Goncharov, Tatiana Varfolomeev, Eugene Xie, Luke Ngu, Hai Peng, Ivan Anderson, Keith R. Verschueren, Erik Choi, Meena Kirkpatrick, Donald S. Easton, Amy Webster, Joshua D. McKenzie, Brent S. Vucic, Domagoj Bingol, Baris Cell Death Dis Article RIP1 kinase-mediated inflammatory and cell death pathways have been implicated in the pathology of acute and chronic disorders of the nervous system. Here, we describe a novel animal model of RIP1 kinase deficiency, generated by knock-in of the kinase-inactivating RIP1(D138N) mutation in rats. Homozygous RIP1 kinase-dead (KD) rats had normal development, reproduction and did not show any gross phenotypes at baseline. However, cells derived from RIP1 KD rats displayed resistance to necroptotic cell death. In addition, RIP1 KD rats were resistant to TNF-induced systemic shock. We studied the utility of RIP1 KD rats for neurological disorders by testing the efficacy of the genetic inactivation in the transient middle cerebral artery occlusion/reperfusion model of brain injury. RIP1 KD rats were protected in this model in a battery of behavioral, imaging, and histopathological endpoints. In addition, RIP1 KD rats had reduced inflammation and accumulation of neuronal injury biomarkers. Unbiased proteomics in the plasma identified additional changes that were ameliorated by RIP1 genetic inactivation. Together these data highlight the utility of the RIP1 KD rats for target validation and biomarker studies for neurological disorders. Nature Publishing Group UK 2021-04-07 /pmc/articles/PMC8026634/ /pubmed/33828080 http://dx.doi.org/10.1038/s41419-021-03651-6 Text en © The Author(s) 2021 Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons license and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/.
spellingShingle Article
Stark, Kimberly
Goncharov, Tatiana
Varfolomeev, Eugene
Xie, Luke
Ngu, Hai
Peng, Ivan
Anderson, Keith R.
Verschueren, Erik
Choi, Meena
Kirkpatrick, Donald S.
Easton, Amy
Webster, Joshua D.
McKenzie, Brent S.
Vucic, Domagoj
Bingol, Baris
Genetic inactivation of RIP1 kinase activity in rats protects against ischemic brain injury
title Genetic inactivation of RIP1 kinase activity in rats protects against ischemic brain injury
title_full Genetic inactivation of RIP1 kinase activity in rats protects against ischemic brain injury
title_fullStr Genetic inactivation of RIP1 kinase activity in rats protects against ischemic brain injury
title_full_unstemmed Genetic inactivation of RIP1 kinase activity in rats protects against ischemic brain injury
title_short Genetic inactivation of RIP1 kinase activity in rats protects against ischemic brain injury
title_sort genetic inactivation of rip1 kinase activity in rats protects against ischemic brain injury
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8026634/
https://www.ncbi.nlm.nih.gov/pubmed/33828080
http://dx.doi.org/10.1038/s41419-021-03651-6
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