Polydatin and Nicotinamide Prevent Iron Accumulation and Lipid Peroxidation in Cellular Models of Mitochondrial Diseases

Ferroptosis, an iron-dependent form of non-apoptotic cell death, is regulated by a complex network involving lipid metabolism, iron homeostasis, and the oxidative-reductive system, with iron accumulation and lipid peroxidation as key drivers. Mitochondrial dysfunction and ROS overproduction often un...

Full description

Saved in:
Bibliographic Details
Main Authors: Paula Cilleros-Holgado, David Gómez-Fernández, Rocío Piñero-Pérez, José Manuel Romero-Domínguez, Diana Reche-López, Mónica Álvarez-Córdoba, Ana Romero-González, Alejandra López-Cabrera, Marta Castro De Oliveira, Andrés Rodríguez-Sacristán, Susana González-Granero, José Manuel García-Verdugo, José Antonio Sánchez-Alcázar
Format: Article
Language:English
Published: MDPI AG 2025-02-01
Series:Antioxidants
Subjects:
Online Access:https://www.mdpi.com/2076-3921/14/2/215
Tags: Add Tag
No Tags, Be the first to tag this record!
_version_ 1849723076338515968
author Paula Cilleros-Holgado
David Gómez-Fernández
Rocío Piñero-Pérez
José Manuel Romero-Domínguez
Diana Reche-López
Mónica Álvarez-Córdoba
Ana Romero-González
Alejandra López-Cabrera
Marta Castro De Oliveira
Andrés Rodríguez-Sacristán
Susana González-Granero
José Manuel García-Verdugo
José Antonio Sánchez-Alcázar
author_facet Paula Cilleros-Holgado
David Gómez-Fernández
Rocío Piñero-Pérez
José Manuel Romero-Domínguez
Diana Reche-López
Mónica Álvarez-Córdoba
Ana Romero-González
Alejandra López-Cabrera
Marta Castro De Oliveira
Andrés Rodríguez-Sacristán
Susana González-Granero
José Manuel García-Verdugo
José Antonio Sánchez-Alcázar
author_sort Paula Cilleros-Holgado
collection DOAJ
description Ferroptosis, an iron-dependent form of non-apoptotic cell death, is regulated by a complex network involving lipid metabolism, iron homeostasis, and the oxidative-reductive system, with iron accumulation and lipid peroxidation as key drivers. Mitochondrial dysfunction and ROS overproduction often underlie the pathogenesis of mitochondrial diseases, for which treatment options are limited, emphasizing the need for novel therapies. In this study, we investigated whether polydatin and nicotinamide could reverse ferroptosis-related pathological features in cellular models derived from patients with pathogenic <i>GFM1</i> variants. Mutant fibroblasts showed increased iron and lipofuscin accumulation, altered expression of iron metabolism-related proteins, elevated lipid peroxidation, and heightened susceptibility to erastin-induced ferroptosis. Treatment with polydatin and nicotinamide effectively corrected these alterations and reduced iron accumulation and lipid peroxidation in induced neurons. Furthermore, chloramphenicol treatment in control cells mimicked the mutant phenotype, suggesting that these pathological changes are linked to the mitochondrial protein synthesis defect characteristic of pathogenic <i>GFM1</i> variants. Notably, adding vitamin E to the polydatin and nicotinamide co-treatment resulted in a reduction in the minimum effective concentration, suggesting potential benefits of its inclusion. In conclusion, the combination of polydatin, nicotinamide, and vitamin E could represent a promising therapeutic option for patients with mitochondrial disorders caused by pathogenic <i>GFM1</i> variants.
format Article
id doaj-art-e6b305f547bf4c388bca1d1e43416a4e
institution DOAJ
issn 2076-3921
language English
publishDate 2025-02-01
publisher MDPI AG
record_format Article
series Antioxidants
spelling doaj-art-e6b305f547bf4c388bca1d1e43416a4e2025-08-20T03:11:07ZengMDPI AGAntioxidants2076-39212025-02-0114221510.3390/antiox14020215Polydatin and Nicotinamide Prevent Iron Accumulation and Lipid Peroxidation in Cellular Models of Mitochondrial DiseasesPaula Cilleros-Holgado0David Gómez-Fernández1Rocío Piñero-Pérez2José Manuel Romero-Domínguez3Diana Reche-López4Mónica Álvarez-Córdoba5Ana Romero-González6Alejandra López-Cabrera7Marta Castro De Oliveira8Andrés Rodríguez-Sacristán9Susana González-Granero10José Manuel García-Verdugo11José Antonio Sánchez-Alcázar12Centro Andaluz de Biología del Desarrollo (CABD-CSIC-Universidad Pablo de Olavide), 41013 Sevilla, SpainCentro Andaluz de Biología del Desarrollo (CABD-CSIC-Universidad Pablo de Olavide), 41013 Sevilla, SpainCentro Andaluz de Biología del Desarrollo (CABD-CSIC-Universidad Pablo de Olavide), 41013 Sevilla, SpainCentro Andaluz de Biología del Desarrollo (CABD-CSIC-Universidad Pablo de Olavide), 41013 Sevilla, SpainCentro Andaluz de Biología del Desarrollo (CABD-CSIC-Universidad Pablo de Olavide), 41013 Sevilla, SpainCentro Andaluz de Biología del Desarrollo (CABD-CSIC-Universidad Pablo de Olavide), 41013 Sevilla, SpainCentro Andaluz de Biología del Desarrollo (CABD-CSIC-Universidad Pablo de Olavide), 41013 Sevilla, SpainCentro Andaluz de Biología del Desarrollo (CABD-CSIC-Universidad Pablo de Olavide), 41013 Sevilla, SpainNeuropediatria, Neurolinkia, C. Jardín de la Isla, 8, Local 4 y 5, 41014 Sevilla, SpainNeuropediatría, Servicio de Pediatría, Hospital Universitario Virgen Macarena, 41009 Sevilla, SpainLaboratory of Comparative Neurobiology, Cavanilles Institute of Biodiversity and Evolutionary Biology, University of Valencia and CIBERNED-ISCIII, 46980 Valencia, SpainLaboratory of Comparative Neurobiology, Cavanilles Institute of Biodiversity and Evolutionary Biology, University of Valencia and CIBERNED-ISCIII, 46980 Valencia, SpainCentro Andaluz de Biología del Desarrollo (CABD-CSIC-Universidad Pablo de Olavide), 41013 Sevilla, SpainFerroptosis, an iron-dependent form of non-apoptotic cell death, is regulated by a complex network involving lipid metabolism, iron homeostasis, and the oxidative-reductive system, with iron accumulation and lipid peroxidation as key drivers. Mitochondrial dysfunction and ROS overproduction often underlie the pathogenesis of mitochondrial diseases, for which treatment options are limited, emphasizing the need for novel therapies. In this study, we investigated whether polydatin and nicotinamide could reverse ferroptosis-related pathological features in cellular models derived from patients with pathogenic <i>GFM1</i> variants. Mutant fibroblasts showed increased iron and lipofuscin accumulation, altered expression of iron metabolism-related proteins, elevated lipid peroxidation, and heightened susceptibility to erastin-induced ferroptosis. Treatment with polydatin and nicotinamide effectively corrected these alterations and reduced iron accumulation and lipid peroxidation in induced neurons. Furthermore, chloramphenicol treatment in control cells mimicked the mutant phenotype, suggesting that these pathological changes are linked to the mitochondrial protein synthesis defect characteristic of pathogenic <i>GFM1</i> variants. Notably, adding vitamin E to the polydatin and nicotinamide co-treatment resulted in a reduction in the minimum effective concentration, suggesting potential benefits of its inclusion. In conclusion, the combination of polydatin, nicotinamide, and vitamin E could represent a promising therapeutic option for patients with mitochondrial disorders caused by pathogenic <i>GFM1</i> variants.https://www.mdpi.com/2076-3921/14/2/215mitochondrial diseases<i>GFM1</i>iron accumulationlipid peroxidationferroptosismtUPR
spellingShingle Paula Cilleros-Holgado
David Gómez-Fernández
Rocío Piñero-Pérez
José Manuel Romero-Domínguez
Diana Reche-López
Mónica Álvarez-Córdoba
Ana Romero-González
Alejandra López-Cabrera
Marta Castro De Oliveira
Andrés Rodríguez-Sacristán
Susana González-Granero
José Manuel García-Verdugo
José Antonio Sánchez-Alcázar
Polydatin and Nicotinamide Prevent Iron Accumulation and Lipid Peroxidation in Cellular Models of Mitochondrial Diseases
Antioxidants
mitochondrial diseases
<i>GFM1</i>
iron accumulation
lipid peroxidation
ferroptosis
mtUPR
title Polydatin and Nicotinamide Prevent Iron Accumulation and Lipid Peroxidation in Cellular Models of Mitochondrial Diseases
title_full Polydatin and Nicotinamide Prevent Iron Accumulation and Lipid Peroxidation in Cellular Models of Mitochondrial Diseases
title_fullStr Polydatin and Nicotinamide Prevent Iron Accumulation and Lipid Peroxidation in Cellular Models of Mitochondrial Diseases
title_full_unstemmed Polydatin and Nicotinamide Prevent Iron Accumulation and Lipid Peroxidation in Cellular Models of Mitochondrial Diseases
title_short Polydatin and Nicotinamide Prevent Iron Accumulation and Lipid Peroxidation in Cellular Models of Mitochondrial Diseases
title_sort polydatin and nicotinamide prevent iron accumulation and lipid peroxidation in cellular models of mitochondrial diseases
topic mitochondrial diseases
<i>GFM1</i>
iron accumulation
lipid peroxidation
ferroptosis
mtUPR
url https://www.mdpi.com/2076-3921/14/2/215
work_keys_str_mv AT paulacillerosholgado polydatinandnicotinamidepreventironaccumulationandlipidperoxidationincellularmodelsofmitochondrialdiseases
AT davidgomezfernandez polydatinandnicotinamidepreventironaccumulationandlipidperoxidationincellularmodelsofmitochondrialdiseases
AT rociopineroperez polydatinandnicotinamidepreventironaccumulationandlipidperoxidationincellularmodelsofmitochondrialdiseases
AT josemanuelromerodominguez polydatinandnicotinamidepreventironaccumulationandlipidperoxidationincellularmodelsofmitochondrialdiseases
AT dianarechelopez polydatinandnicotinamidepreventironaccumulationandlipidperoxidationincellularmodelsofmitochondrialdiseases
AT monicaalvarezcordoba polydatinandnicotinamidepreventironaccumulationandlipidperoxidationincellularmodelsofmitochondrialdiseases
AT anaromerogonzalez polydatinandnicotinamidepreventironaccumulationandlipidperoxidationincellularmodelsofmitochondrialdiseases
AT alejandralopezcabrera polydatinandnicotinamidepreventironaccumulationandlipidperoxidationincellularmodelsofmitochondrialdiseases
AT martacastrodeoliveira polydatinandnicotinamidepreventironaccumulationandlipidperoxidationincellularmodelsofmitochondrialdiseases
AT andresrodriguezsacristan polydatinandnicotinamidepreventironaccumulationandlipidperoxidationincellularmodelsofmitochondrialdiseases
AT susanagonzalezgranero polydatinandnicotinamidepreventironaccumulationandlipidperoxidationincellularmodelsofmitochondrialdiseases
AT josemanuelgarciaverdugo polydatinandnicotinamidepreventironaccumulationandlipidperoxidationincellularmodelsofmitochondrialdiseases
AT joseantoniosanchezalcazar polydatinandnicotinamidepreventironaccumulationandlipidperoxidationincellularmodelsofmitochondrialdiseases