نتایج جستجو برای: mitochondrial encephalomyopathy

تعداد نتایج: 132426  

Journal: :The Journal of biological chemistry 2006
Giovanna Pontarin Paola Ferraro Maria L Valentino Michio Hirano Peter Reichard Vera Bianchi

Mitochondrial (mt) neurogastrointestinal encephalomyopathy (MNGIE) is an autosomal recessive disease associated with depletion, deletions, and point mutations of mtDNA. Patients lack a functional thymidine phosphorylase and their plasma contains high concentrations of thymidine and deoxyuridine; elevation of the corresponding triphosphates probably impairs normal mtDNA replication and repair. T...

Journal: :The Journal of biological chemistry 2005
Paola Ferraro Giovanna Pontarin Laura Crocco Sonia Fabris Peter Reichard Vera Bianchi

Mitochondrial (mt) DNA depletion syndromes can arise from genetic deficiencies for enzymes of dNTP metabolism, operating either inside or outside mitochondria. MNGIE is caused by the deficiency of cytosolic thymidine phosphorylase that degrades thymidine and deoxyuridine. The extracellular fluid of the patients contains 10-20 microM deoxynucleosides leading to changes in dTTP that may disturb m...

2017
Mauro Scarpelli Alice Todeschini Irene Volonghi Alessandro Padovani Massimiliano Filosto

Mitochondrial diseases (MDs) are a clinically heterogeneous group of disorders caused by a dysfunction of the mitochondrial respiratory chain. They can be related to mutation of genes encoded using either nuclear DNA or mitochondrial DNA. The advent of next generation sequencing and whole exome sequencing in studying the molecular bases of MDs will bring about a revolution in the field of mitoc...

Journal: :Brain : a journal of neurology 2007
Rosalba Carrozzo Carlo Dionisi-Vici Ulrike Steuerwald Simona Lucioli Federica Deodato Sivia Di Giandomenico Enrico Bertini Barbara Franke Leo A J Kluijtmans Maria Chiara Meschini Cristiano Rizzo Fiorella Piemonte Richard Rodenburg René Santer Filippo M Santorelli Arno van Rooij Diana Vermunt-de Koning Eva Morava Ron A Wevers

One pedigree with four patients has been recently described with mitochondrial DNA depletion and mutation in SUCLA2 gene leading to succinyl-CoA synthase deficiency. Patients had a Leigh-like encephalomyopathy and deafness but besides the presence of lactic acidosis, the profile of urine organic acid was not reported. We have studied 14 patients with mild 'unlabelled' methylmalonic aciduria (MM...

Journal: :Molecular and cellular biology 1994
M Yoneda T Miyatake G Attardi

The rules that govern complementation of mutant and wild-type mitochondrial genomes in human cells were investigated under different experimental conditions. Among mitochondrial transformants derived from an individual affected by the MERRF (myoclonus epilepsy associated with ragged red fibers) encephalomyopathy and carrying in heteroplasmic form the mitochondrial tRNA(Lys) mutation associated ...

Journal: :Human molecular genetics 2015
Claudia Nesti Maria Chiara Meschini Brigitte Meunier Michele Sacchini Stefano Doccini Alessandro Romano Sara Petrillo Ilaria Pezzini Nadir Seddiki Anna Rubegni Fiorella Piemonte M Alice Donati Gael Brasseur Filippo M Santorelli

We describe the case of a woman in whom combination of a mitochondrial (MT-CYB) and a nuclear (SDHB) mutation was associated with clinical and metabolic features suggestive of a mitochondrial disorder. The mutations impaired overall energy metabolism in the patient's muscle and fibroblasts and increased cellular susceptibility to oxidative stress. To clarify the contribution of each mutation to...

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