EPRS1-mediated fibroblast activation and mitochondrial dysfunction promote kidney fibrosis

Abstract Kidney fibrosis causes irreversible structural damage in chronic kidney disease and is characterized by aberrant extracellular matrix (ECM) accumulation. Although glutamyl-prolyl-tRNA synthetase 1 (EPRS1) is a crucial enzyme involved in proline-rich protein synthesis, its role in kidney fib...

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Main Authors: Seung Seob Son, Hee Seul Jeong, Seong-Woo Lee, Eun Soo Lee, Jeong Geon Lee, Ji-Hye Lee, Jawoon Yi, Mi Ju Park, Min Sun Choi, Donghyeong Lee, Sin Young Choi, Jiheon Ha, Jeong Suk Kang, Nam-Jun Cho, Samel Park, Hyo-Wook Gil, Choon Hee Chung, Joon Seok Park, Myung Hee Kim, Jihwan Park, Eun Young Lee
Format: Article
Language:English
Published: Nature Publishing Group 2024-12-01
Series:Experimental and Molecular Medicine
Online Access:https://doi.org/10.1038/s12276-024-01360-6
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author Seung Seob Son
Hee Seul Jeong
Seong-Woo Lee
Eun Soo Lee
Jeong Geon Lee
Ji-Hye Lee
Jawoon Yi
Mi Ju Park
Min Sun Choi
Donghyeong Lee
Sin Young Choi
Jiheon Ha
Jeong Suk Kang
Nam-Jun Cho
Samel Park
Hyo-Wook Gil
Choon Hee Chung
Joon Seok Park
Myung Hee Kim
Jihwan Park
Eun Young Lee
author_facet Seung Seob Son
Hee Seul Jeong
Seong-Woo Lee
Eun Soo Lee
Jeong Geon Lee
Ji-Hye Lee
Jawoon Yi
Mi Ju Park
Min Sun Choi
Donghyeong Lee
Sin Young Choi
Jiheon Ha
Jeong Suk Kang
Nam-Jun Cho
Samel Park
Hyo-Wook Gil
Choon Hee Chung
Joon Seok Park
Myung Hee Kim
Jihwan Park
Eun Young Lee
author_sort Seung Seob Son
collection DOAJ
description Abstract Kidney fibrosis causes irreversible structural damage in chronic kidney disease and is characterized by aberrant extracellular matrix (ECM) accumulation. Although glutamyl-prolyl-tRNA synthetase 1 (EPRS1) is a crucial enzyme involved in proline-rich protein synthesis, its role in kidney fibrosis remains unclear. The present study revealed that EPRS1 expression levels were increased in the fibrotic kidneys of patients and mice, especially in fibroblasts and proximal tubular epithelial cells, on the basis of single-cell analysis and immunostaining of fibrotic kidneys. Moreover, C57BL/6 EPRS1tm1b heterozygous knockout (Eprs1 +/−) and pharmacological EPRS1 inhibition with the first-in-class EPRS1 inhibitor DWN12088 protected against kidney fibrosis and dysfunction by preventing fibroblast activation and proximal tubular injury. Interestingly, in vitro assays demonstrated that EPRS1-mediated nontranslational pathways in addition to translational pathways under transforming growth factor β-treated conditions by phosphorylating SMAD family member 3 in fibroblasts and signal transducers and activators of transcription 3 in injured proximal tubules. EPRS1 knockdown and catalytic inhibition suppressed these pathways, preventing fibroblast activation, proliferation, and subsequent collagen production. Additionally, we revealed that EPRS1 caused mitochondrial damage in proximal tubules but that this damage was attenuated by EPRS1 inhibition. Our findings suggest that the EPRS1-mediated ECM accumulation induces kidney fibrosis via fibroblast activation and mitochondrial dysfunction. Therefore, targeting EPRS1 could be a potential therapeutic target for alleviating fibrotic injury in chronic kidney disease.
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spelling doaj-art-299c481027ec4cea9d2eeca85fa028b22025-08-20T02:43:24ZengNature Publishing GroupExperimental and Molecular Medicine2092-64132024-12-0156122673268910.1038/s12276-024-01360-6EPRS1-mediated fibroblast activation and mitochondrial dysfunction promote kidney fibrosisSeung Seob Son0Hee Seul Jeong1Seong-Woo Lee2Eun Soo Lee3Jeong Geon Lee4Ji-Hye Lee5Jawoon Yi6Mi Ju Park7Min Sun Choi8Donghyeong Lee9Sin Young Choi10Jiheon Ha11Jeong Suk Kang12Nam-Jun Cho13Samel Park14Hyo-Wook Gil15Choon Hee Chung16Joon Seok Park17Myung Hee Kim18Jihwan Park19Eun Young Lee20Department of Medicine, Graduate School of Soonchunhyang UniversityDepartment of Medicine, Graduate School of Soonchunhyang UniversityDepartment of Medicine, Graduate School of Soonchunhyang UniversityDepartment of Internal Medicine, Yonsei University Wonju College of MedicineDepartment of Medicine, College of Medicine, Soonchunhyang UniversityDepartment of Medicine, College of Medicine, Soonchunhyang UniversitySchool of Life Sciences, Gwangju Institute of Science and Technology (GIST)Department of Medicine, Graduate School of Soonchunhyang UniversityDepartment of Medicine, Graduate School of Soonchunhyang UniversityDepartment of Medicine, Graduate School of Soonchunhyang UniversitySchool of Life Sciences, Gwangju Institute of Science and Technology (GIST)Department of Medicine, College of Medicine, Soonchunhyang UniversityDepartment of Internal Medicine, Soonchunhyang University Cheonan HospitalDepartment of Medicine, College of Medicine, Soonchunhyang UniversityDepartment of Medicine, College of Medicine, Soonchunhyang UniversityDepartment of Medicine, College of Medicine, Soonchunhyang UniversityDepartment of Internal Medicine, Yonsei University Wonju College of MedicineDrug Discovery Center, Daewoong Pharmaceutical Co. Ltd.Microbiome Convergence Research Center, Korea Research Institute of Bioscience and BiotechnologySchool of Life Sciences, Gwangju Institute of Science and Technology (GIST)Department of Medicine, Graduate School of Soonchunhyang UniversityAbstract Kidney fibrosis causes irreversible structural damage in chronic kidney disease and is characterized by aberrant extracellular matrix (ECM) accumulation. Although glutamyl-prolyl-tRNA synthetase 1 (EPRS1) is a crucial enzyme involved in proline-rich protein synthesis, its role in kidney fibrosis remains unclear. The present study revealed that EPRS1 expression levels were increased in the fibrotic kidneys of patients and mice, especially in fibroblasts and proximal tubular epithelial cells, on the basis of single-cell analysis and immunostaining of fibrotic kidneys. Moreover, C57BL/6 EPRS1tm1b heterozygous knockout (Eprs1 +/−) and pharmacological EPRS1 inhibition with the first-in-class EPRS1 inhibitor DWN12088 protected against kidney fibrosis and dysfunction by preventing fibroblast activation and proximal tubular injury. Interestingly, in vitro assays demonstrated that EPRS1-mediated nontranslational pathways in addition to translational pathways under transforming growth factor β-treated conditions by phosphorylating SMAD family member 3 in fibroblasts and signal transducers and activators of transcription 3 in injured proximal tubules. EPRS1 knockdown and catalytic inhibition suppressed these pathways, preventing fibroblast activation, proliferation, and subsequent collagen production. Additionally, we revealed that EPRS1 caused mitochondrial damage in proximal tubules but that this damage was attenuated by EPRS1 inhibition. Our findings suggest that the EPRS1-mediated ECM accumulation induces kidney fibrosis via fibroblast activation and mitochondrial dysfunction. Therefore, targeting EPRS1 could be a potential therapeutic target for alleviating fibrotic injury in chronic kidney disease.https://doi.org/10.1038/s12276-024-01360-6
spellingShingle Seung Seob Son
Hee Seul Jeong
Seong-Woo Lee
Eun Soo Lee
Jeong Geon Lee
Ji-Hye Lee
Jawoon Yi
Mi Ju Park
Min Sun Choi
Donghyeong Lee
Sin Young Choi
Jiheon Ha
Jeong Suk Kang
Nam-Jun Cho
Samel Park
Hyo-Wook Gil
Choon Hee Chung
Joon Seok Park
Myung Hee Kim
Jihwan Park
Eun Young Lee
EPRS1-mediated fibroblast activation and mitochondrial dysfunction promote kidney fibrosis
Experimental and Molecular Medicine
title EPRS1-mediated fibroblast activation and mitochondrial dysfunction promote kidney fibrosis
title_full EPRS1-mediated fibroblast activation and mitochondrial dysfunction promote kidney fibrosis
title_fullStr EPRS1-mediated fibroblast activation and mitochondrial dysfunction promote kidney fibrosis
title_full_unstemmed EPRS1-mediated fibroblast activation and mitochondrial dysfunction promote kidney fibrosis
title_short EPRS1-mediated fibroblast activation and mitochondrial dysfunction promote kidney fibrosis
title_sort eprs1 mediated fibroblast activation and mitochondrial dysfunction promote kidney fibrosis
url https://doi.org/10.1038/s12276-024-01360-6
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