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Genome-wide association studies identify the role of caspase-9 in kidney disease

Genome-wide association studies (GWAS) have identified hundreds of genetic risk regions for kidney dysfunction [estimated glomerular filtration rate (eGFR)]; however, the causal genes, cell types, and pathways are poorly understood. Integration of GWAS and human kidney expression of quantitative tra...

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Autores principales: Doke, Tomohito, Huang, Shizheng, Qiu, Chengxiang, Sheng, Xin, Seasock, Matthew, Liu, Hongbo, Ma, Ziyuan, Palmer, Matthew, Susztak, Katalin
Formato: Online Artículo Texto
Lenguaje:English
Publicado: American Association for the Advancement of Science 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8570608/
https://www.ncbi.nlm.nih.gov/pubmed/34739325
http://dx.doi.org/10.1126/sciadv.abi8051
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author Doke, Tomohito
Huang, Shizheng
Qiu, Chengxiang
Sheng, Xin
Seasock, Matthew
Liu, Hongbo
Ma, Ziyuan
Palmer, Matthew
Susztak, Katalin
author_facet Doke, Tomohito
Huang, Shizheng
Qiu, Chengxiang
Sheng, Xin
Seasock, Matthew
Liu, Hongbo
Ma, Ziyuan
Palmer, Matthew
Susztak, Katalin
author_sort Doke, Tomohito
collection PubMed
description Genome-wide association studies (GWAS) have identified hundreds of genetic risk regions for kidney dysfunction [estimated glomerular filtration rate (eGFR)]; however, the causal genes, cell types, and pathways are poorly understood. Integration of GWAS and human kidney expression of quantitative trait analysis using Bayesian colocations, transcriptome-wide association studies, and summary-based Mendelian randomization studies prioritized caspase-9 (CASP9) as a kidney disease risk gene. Human kidney single-cell epigenetic and immunostaining studies indicated kidney tubule cells as a disease-causing cell type. Mice with genetic deletion or pharmacological inhibition of CASP9 showed lower apoptosis while having improved mitophagy, resulting in dampened activation of cytosolic nucleotide sensing pathways (cGAS-STING), reduction of inflammation, and protection from acute kidney disease or renal fibrosis. In summary, here, we prioritized CASP9 as an eGFR GWAS target gene and demonstrated the causal role of CASP9 in kidney disease development via improving mitophagy and lowering inflammation and apoptosis.
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spelling pubmed-85706082021-11-17 Genome-wide association studies identify the role of caspase-9 in kidney disease Doke, Tomohito Huang, Shizheng Qiu, Chengxiang Sheng, Xin Seasock, Matthew Liu, Hongbo Ma, Ziyuan Palmer, Matthew Susztak, Katalin Sci Adv Biomedicine and Life Sciences Genome-wide association studies (GWAS) have identified hundreds of genetic risk regions for kidney dysfunction [estimated glomerular filtration rate (eGFR)]; however, the causal genes, cell types, and pathways are poorly understood. Integration of GWAS and human kidney expression of quantitative trait analysis using Bayesian colocations, transcriptome-wide association studies, and summary-based Mendelian randomization studies prioritized caspase-9 (CASP9) as a kidney disease risk gene. Human kidney single-cell epigenetic and immunostaining studies indicated kidney tubule cells as a disease-causing cell type. Mice with genetic deletion or pharmacological inhibition of CASP9 showed lower apoptosis while having improved mitophagy, resulting in dampened activation of cytosolic nucleotide sensing pathways (cGAS-STING), reduction of inflammation, and protection from acute kidney disease or renal fibrosis. In summary, here, we prioritized CASP9 as an eGFR GWAS target gene and demonstrated the causal role of CASP9 in kidney disease development via improving mitophagy and lowering inflammation and apoptosis. American Association for the Advancement of Science 2021-11-05 /pmc/articles/PMC8570608/ /pubmed/34739325 http://dx.doi.org/10.1126/sciadv.abi8051 Text en Copyright © 2021 The Authors, some rights reserved; exclusive licensee American Association for the Advancement of Science. No claim to original U.S. Government Works. Distributed under a Creative Commons Attribution License 4.0 (CC BY). https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution license (https://creativecommons.org/licenses/by/4.0/) , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Biomedicine and Life Sciences
Doke, Tomohito
Huang, Shizheng
Qiu, Chengxiang
Sheng, Xin
Seasock, Matthew
Liu, Hongbo
Ma, Ziyuan
Palmer, Matthew
Susztak, Katalin
Genome-wide association studies identify the role of caspase-9 in kidney disease
title Genome-wide association studies identify the role of caspase-9 in kidney disease
title_full Genome-wide association studies identify the role of caspase-9 in kidney disease
title_fullStr Genome-wide association studies identify the role of caspase-9 in kidney disease
title_full_unstemmed Genome-wide association studies identify the role of caspase-9 in kidney disease
title_short Genome-wide association studies identify the role of caspase-9 in kidney disease
title_sort genome-wide association studies identify the role of caspase-9 in kidney disease
topic Biomedicine and Life Sciences
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8570608/
https://www.ncbi.nlm.nih.gov/pubmed/34739325
http://dx.doi.org/10.1126/sciadv.abi8051
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