Mechanotransductive stabilization of HIF-1α is inhibited by mitochondrial antioxidant therapy in the setting of pulmonary overcirculation

Abstract In patients with congenital heart disease, the development of pulmonary arterial hypertension (PAH) is based on vascular exposure to abnormal hemodynamic forces. In our work using a large animal model of increased pulmonary blood flow and pressure, we have previously described a pattern of...

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Main Authors: Jason T. Boehme, Sanjeev A. Datar, Xutong Sun, Wenhui Gong, Qing Lu, Jamie Soto, Michael A. Smith, Alejandro E. Garcia-Flores, Gary W. Raff, Ting Wang, Emin Maltepe, Stephen M. Black, Jeffrey R. Fineman
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Language:English
Published: Nature Portfolio 2025-05-01
Series:Scientific Reports
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Online Access:https://doi.org/10.1038/s41598-025-99062-0
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author Jason T. Boehme
Sanjeev A. Datar
Xutong Sun
Wenhui Gong
Qing Lu
Jamie Soto
Michael A. Smith
Alejandro E. Garcia-Flores
Gary W. Raff
Ting Wang
Emin Maltepe
Stephen M. Black
Jeffrey R. Fineman
author_facet Jason T. Boehme
Sanjeev A. Datar
Xutong Sun
Wenhui Gong
Qing Lu
Jamie Soto
Michael A. Smith
Alejandro E. Garcia-Flores
Gary W. Raff
Ting Wang
Emin Maltepe
Stephen M. Black
Jeffrey R. Fineman
author_sort Jason T. Boehme
collection DOAJ
description Abstract In patients with congenital heart disease, the development of pulmonary arterial hypertension (PAH) is based on vascular exposure to abnormal hemodynamic forces. In our work using a large animal model of increased pulmonary blood flow and pressure, we have previously described a pattern of alterations to vascular cell metabolism, mitochondrial function, and mitochondrial redox signaling, paralleling changes in advanced pulmonary vasculopathy states. Based on our findings and emerging literature, we believe that endothelial mitochondria play a central role in integrating and relaying pathologic mechanotransductive signals in abnormal pulmonary hemodynamics. In this manuscript, we demonstrate that exposure of the pulmonary vascular endothelium to aberrant mechanical forces increases production of mitochondrial reactive oxygen species (ROS) and stabilizes the transcription factor Hypoxia Inducible Factor-1α (HIF-1α), and that these changes are associated with impaired endothelial production of Nitric Oxide (NO). We validate that the mitochondrial antioxidant 10-(6′-ubiquinonyl)decyltriphenylphosphonium bromide (MitoQ) can reverse these alterations in vitro, and evaluate the effects of MitoQ treatment in vivo utilizing our large animal shunt model. We find that MitoQ therapy in pulmonary overcirculation decreases the production of mitochondrial ROS, diminishes the mechanically-induced stabilization of HIF-1α, and partially restores vascular reactivity by rescuing endothelial NO production. These findings raise exciting prospects concerning shared pathophysiologic mechanisms and possible common therapeutic targets amongst PAH etiologies.
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spelling doaj-art-a945809dd00548d2b65c984fa56296fa2025-08-20T03:09:35ZengNature PortfolioScientific Reports2045-23222025-05-0115111510.1038/s41598-025-99062-0Mechanotransductive stabilization of HIF-1α is inhibited by mitochondrial antioxidant therapy in the setting of pulmonary overcirculationJason T. Boehme0Sanjeev A. Datar1Xutong Sun2Wenhui Gong3Qing Lu4Jamie Soto5Michael A. Smith6Alejandro E. Garcia-Flores7Gary W. Raff8Ting Wang9Emin Maltepe10Stephen M. Black11Jeffrey R. Fineman12Department of Pediatrics, University of California San FranciscoDepartment of Pediatrics, University of California San FranciscoCenter for Translational Science, Florida International UniversityDepartment of Pediatrics, University of California San FranciscoCenter for Translational Science, Florida International UniversityCenter for Translational Science, Florida International UniversityDepartment of Pediatrics, University of California San FranciscoCenter for Translational Science, Florida International UniversityDepartment of Surgery, University of California DavisCenter for Translational Science, Florida International UniversityDepartment of Pediatrics, University of California San FranciscoCenter for Translational Science, Florida International UniversityDepartment of Pediatrics, University of California San FranciscoAbstract In patients with congenital heart disease, the development of pulmonary arterial hypertension (PAH) is based on vascular exposure to abnormal hemodynamic forces. In our work using a large animal model of increased pulmonary blood flow and pressure, we have previously described a pattern of alterations to vascular cell metabolism, mitochondrial function, and mitochondrial redox signaling, paralleling changes in advanced pulmonary vasculopathy states. Based on our findings and emerging literature, we believe that endothelial mitochondria play a central role in integrating and relaying pathologic mechanotransductive signals in abnormal pulmonary hemodynamics. In this manuscript, we demonstrate that exposure of the pulmonary vascular endothelium to aberrant mechanical forces increases production of mitochondrial reactive oxygen species (ROS) and stabilizes the transcription factor Hypoxia Inducible Factor-1α (HIF-1α), and that these changes are associated with impaired endothelial production of Nitric Oxide (NO). We validate that the mitochondrial antioxidant 10-(6′-ubiquinonyl)decyltriphenylphosphonium bromide (MitoQ) can reverse these alterations in vitro, and evaluate the effects of MitoQ treatment in vivo utilizing our large animal shunt model. We find that MitoQ therapy in pulmonary overcirculation decreases the production of mitochondrial ROS, diminishes the mechanically-induced stabilization of HIF-1α, and partially restores vascular reactivity by rescuing endothelial NO production. These findings raise exciting prospects concerning shared pathophysiologic mechanisms and possible common therapeutic targets amongst PAH etiologies.https://doi.org/10.1038/s41598-025-99062-0HIF-1αMitochondrial ROSMechanotransductionNitric oxidePulmonary hypertensionCongenital heart disease
spellingShingle Jason T. Boehme
Sanjeev A. Datar
Xutong Sun
Wenhui Gong
Qing Lu
Jamie Soto
Michael A. Smith
Alejandro E. Garcia-Flores
Gary W. Raff
Ting Wang
Emin Maltepe
Stephen M. Black
Jeffrey R. Fineman
Mechanotransductive stabilization of HIF-1α is inhibited by mitochondrial antioxidant therapy in the setting of pulmonary overcirculation
Scientific Reports
HIF-1α
Mitochondrial ROS
Mechanotransduction
Nitric oxide
Pulmonary hypertension
Congenital heart disease
title Mechanotransductive stabilization of HIF-1α is inhibited by mitochondrial antioxidant therapy in the setting of pulmonary overcirculation
title_full Mechanotransductive stabilization of HIF-1α is inhibited by mitochondrial antioxidant therapy in the setting of pulmonary overcirculation
title_fullStr Mechanotransductive stabilization of HIF-1α is inhibited by mitochondrial antioxidant therapy in the setting of pulmonary overcirculation
title_full_unstemmed Mechanotransductive stabilization of HIF-1α is inhibited by mitochondrial antioxidant therapy in the setting of pulmonary overcirculation
title_short Mechanotransductive stabilization of HIF-1α is inhibited by mitochondrial antioxidant therapy in the setting of pulmonary overcirculation
title_sort mechanotransductive stabilization of hif 1α is inhibited by mitochondrial antioxidant therapy in the setting of pulmonary overcirculation
topic HIF-1α
Mitochondrial ROS
Mechanotransduction
Nitric oxide
Pulmonary hypertension
Congenital heart disease
url https://doi.org/10.1038/s41598-025-99062-0
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