Literature DB >> 7603512

Analysis of cybrids harboring MELAS mutations in the mitochondrial tRNA(Leu(UUR)) gene.

Y Koga1, M Davidson, E A Schon, M P King.   

Abstract

MELAS (mitochondrial myopathy, encephalopathy, lactic acidosis, and strokelike episodes), a maternally inherited mitochondrial disorder, has been associated with an A-->G transition at nucleotide 3243 and a T-->C transition at nucleotide 3271, both in the mitochondrial tRNA(Leu(UUR)) gene. We transferred mitochondria harboring these mutations into human cells lacking endogenous mtDNA (rho o cells), and analyzed the resulting transmitochondrial cytoplasmic hybrid (cybrid) cell lines for the relationship of genotype to phenotype. Cybrids containing high levels of mutated genomes showed decreased rates of synthesis of mitochondrial translation products, reduced respiratory chain function, and increased amounts of a novel unprocessed RNA species (RNA 19). Overall effects on mitochondrial functions were more severe for the MELAS 3243 cybrids as compared to the MELAS 3271 cybrids. These data, combined with our previous observations, suggest that RNA 19 may play an important, but as yet uncharacterized, role in the pathogenesis of this mitochondrial disorder.

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Year:  1995        PMID: 7603512     DOI: 10.1002/mus.880181424

Source DB:  PubMed          Journal:  Muscle Nerve Suppl


  8 in total

1.  Normal levels of wild-type mitochondrial DNA maintain cytochrome c oxidase activity for two pathogenic mitochondrial DNA mutations but not for m.3243A-->G.

Authors:  Steve E Durham; David C Samuels; Lynsey M Cree; Patrick F Chinnery
Journal:  Am J Hum Genet       Date:  2007-05-23       Impact factor: 11.025

Review 2.  Mitochondrial DNA mutations and pathogenesis.

Authors:  E A Schon; E Bonilla; S DiMauro
Journal:  J Bioenerg Biomembr       Date:  1997-04       Impact factor: 2.945

3.  Point mutations in the mitochondrial tRNA(Lys) gene: implications for pathogenesis and mechanism.

Authors:  J P Masucci; E A Schon; M P King
Journal:  Mol Cell Biochem       Date:  1997-09       Impact factor: 3.396

4.  Mitochondrial DNA variants can mediate methylation status of inflammation, angiogenesis and signaling genes.

Authors:  Shari R Atilano; Deepika Malik; Marilyn Chwa; Javier Cáceres-Del-Carpio; Anthony B Nesburn; David S Boyer; Baruch D Kuppermann; S Michal Jazwinski; Michael V Miceli; Douglas C Wallace; Nitin Udar; M Cristina Kenney
Journal:  Hum Mol Genet       Date:  2015-05-10       Impact factor: 6.150

5.  Measurement of ATP production in mitochondrial disorders.

Authors:  R K Shepherd; N Checcarelli; A Naini; D C De Vivo; S DiMauro; C M Sue
Journal:  J Inherit Metab Dis       Date:  2006-02       Impact factor: 4.982

6.  Biochemical analysis of respiratory function in cybrid cell lines harbouring mitochondrial DNA mutations.

Authors:  Francesco Pallotti; Alessandra Baracca; Evelyn Hernandez-Rosa; Winsome F Walker; Giancarlo Solaini; Giorgio Lenaz; Gian Vico Melzi D'Eril; Salvatore Dimauro; Eric A Schon; Mercy M Davidson
Journal:  Biochem J       Date:  2004-12-01       Impact factor: 3.857

Review 7.  Cytoplasmic hybrid (cybrid) cell lines as a practical model for mitochondriopathies.

Authors:  Heather M Wilkins; Steven M Carl; Russell H Swerdlow
Journal:  Redox Biol       Date:  2014-04-01       Impact factor: 11.799

8.  A Primer Genetic Toolkit for Exploring Mitochondrial Biology and Disease Using Zebrafish.

Authors:  Ankit Sabharwal; Jarryd M Campbell; Tanya L Schwab; Zachary WareJoncas; Mark D Wishman; Hirotaka Ata; Wiebin Liu; Noriko Ichino; Danielle E Hunter; Jake D Bergren; Mark D Urban; Rhianna M Urban; Shannon R Holmberg; Bibekananda Kar; Alex Cook; Yonghe Ding; Xiaolei Xu; Karl J Clark; Stephen C Ekker
Journal:  Genes (Basel)       Date:  2022-07-23       Impact factor: 4.141

  8 in total

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