Literature DB >> 35684429

Personalized Medicine in Mitochondrial Health and Disease: Molecular Basis of Therapeutic Approaches Based on Nutritional Supplements and Their Analogs.

Vincenzo Tragni1,2, Guido Primiano3,4, Albina Tummolo5, Lucas Cafferati Beltrame1, Gianluigi La Piana1, Maria Noemi Sgobba1, Maria Maddalena Cavalluzzi6, Giulia Paterno5, Ruggiero Gorgoglione1, Mariateresa Volpicella1, Lorenzo Guerra1, Domenico Marzulli2, Serenella Servidei3,4, Anna De Grassi1, Giuseppe Petrosillo2, Giovanni Lentini6, Ciro Leonardo Pierri1.   

Abstract

Mitochondrial diseases (MDs) may result from mutations affecting nuclear or mitochondrial genes, encoding mitochondrial proteins, or non-protein-coding mitochondrial RNA. Despite the great variability of affected genes, in the most severe cases, a neuromuscular and neurodegenerative phenotype is observed, and no specific therapy exists for a complete recovery from the disease. The most used treatments are symptomatic and based on the administration of antioxidant cocktails combined with antiepileptic/antipsychotic drugs and supportive therapy for multiorgan involvement. Nevertheless, the real utility of antioxidant cocktail treatments for patients affected by MDs still needs to be scientifically demonstrated. Unfortunately, clinical trials for antioxidant therapies using α-tocopherol, ascorbate, glutathione, riboflavin, niacin, acetyl-carnitine and coenzyme Q have met a limited success. Indeed, it would be expected that the employed antioxidants can only be effective if they are able to target the specific mechanism, i.e., involving the central and peripheral nervous system, responsible for the clinical manifestations of the disease. Noteworthily, very often the phenotypes characterizing MD patients are associated with mutations in proteins whose function does not depend on specific cofactors. Conversely, the administration of the antioxidant cocktails might determine the suppression of endogenous oxidants resulting in deleterious effects on cell viability and/or toxicity for patients. In order to avoid toxicity effects and before administering the antioxidant therapy, it might be useful to ascertain the blood serum levels of antioxidants and cofactors to be administered in MD patients. It would be also worthwhile to check the localization of mutations affecting proteins whose function should depend (less or more directly) on the cofactors to be administered, for estimating the real need and predicting the success of the proposed cofactor/antioxidant-based therapy.

Entities:  

Keywords:  CRAT deficiency; Leigh syndrome; Leigh-like syndromes; MEGDEL; MELAS; MERRF; SLC25A10 and DIC deficiency; aminoacyl tRNA synthetase; antioxidant cocktails; cofactors; complex I; dietary supplement; encephalomyopathies; mitochondrial carriers; mitochondrial diseases; mitochondrial dysfunction; mitochondrial impairment; peptide-based treatments; phospholipids; type I NADH dehydrogenase; vitamins

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Year:  2022        PMID: 35684429      PMCID: PMC9182050          DOI: 10.3390/molecules27113494

Source DB:  PubMed          Journal:  Molecules        ISSN: 1420-3049            Impact factor:   4.927


  239 in total

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Journal:  Biochim Biophys Acta       Date:  2009-06-17

Review 2.  Mitochondrial tRNA import and its consequences for mitochondrial translation.

Authors:  André Schneider
Journal:  Annu Rev Biochem       Date:  2011       Impact factor: 23.643

3.  Crystal structure of human mitochondrial PheRS complexed with tRNA(Phe) in the active "open" state.

Authors:  Liron Klipcan; Nina Moor; Igal Finarov; Naama Kessler; Maria Sukhanova; Mark G Safro
Journal:  J Mol Biol       Date:  2011-11-23       Impact factor: 5.469

4.  Biotin rescues mitochondrial dysfunction and neurotoxicity in a tauopathy model.

Authors:  Kelly M Lohr; Bess Frost; Clemens Scherzer; Mel B Feany
Journal:  Proc Natl Acad Sci U S A       Date:  2020-12-14       Impact factor: 11.205

5.  Fatal ataxic encephalopathy and carnitine acetyltransferase deficiency: a functional defect of pyruvate oxidation?

Authors:  S DiDonato; M Rimoldi; A Moise; B Bertagnoglio; G Uziel
Journal:  Neurology       Date:  1979-12       Impact factor: 9.910

6.  Cocrystal structures of glycyl-tRNA synthetase in complex with tRNA suggest multiple conformational states in glycylation.

Authors:  Xiangjing Qin; Zhitai Hao; Qingnan Tian; Zhemin Zhang; Chun Zhou; Wei Xie
Journal:  J Biol Chem       Date:  2014-06-04       Impact factor: 5.157

7.  An intramembrane chaperone complex facilitates membrane protein biogenesis.

Authors:  Patrick J Chitwood; Ramanujan S Hegde
Journal:  Nature       Date:  2020-08-19       Impact factor: 49.962

Review 8.  The molecular pathology of pathogenic mitochondrial tRNA variants.

Authors:  Uwe Richter; Robert McFarland; Robert W Taylor; Sarah J Pickett
Journal:  FEBS Lett       Date:  2021-02-12       Impact factor: 3.864

9.  A Leigh syndrome caused by compound heterozygous mutations on NDUFAF5 induce early infant death: A case report.

Authors:  Yan Wen; Guoyan Lu; Lina Qiao; Yifei Li
Journal:  Mol Genet Genomic Med       Date:  2021-12-28       Impact factor: 2.183

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  1 in total

1.  Familial mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episode syndrome: Three case reports.

Authors:  Xiao Yang; Le-Jun Fu
Journal:  World J Clin Cases       Date:  2022-09-26       Impact factor: 1.534

  1 in total

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