Literature DB >> 10716261

Very low levels of the mtDNA A3243G mutation associated with mitochondrial dysfunction in vivo.

P F Chinnery1, D J Taylor, D T Brown, D Manners, P Styles, R Lodi.   

Abstract

We studied mitochondrial function in vivo in 2 brothers harboring the mitochondrial DNA A3243G mutation by using magnetic resonance spectroscopy. One brother presented with recurrent strokes and had a mitochondrial respiratory chain complex I defect, with 85% A3243G mutation in his quadriceps. The maximum rate of mitochondrial ATP production in his calf, measured in vivo, was reduced to 21% of the normal mean value. The second brother had mild exercise intolerance, normal muscle histochemistry, and normal respiratory chain activity in vitro. Despite a level of the A3243G mutation of only 5.95% (SD, 4.45; range, 0.7-16.1%) within single muscle fibers from the gastrocnemius muscle, the maximum rate of mitochondrial ATP production in his calf, measured in vivo, was reduced to 35% of the normal mean value. These findings suggest that there may not be a clear genetic threshold level for the expression of the A3243G mutation in skeletal muscle in vivo.

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Year:  2000        PMID: 10716261

Source DB:  PubMed          Journal:  Ann Neurol        ISSN: 0364-5134            Impact factor:   10.422


  16 in total

1.  Heteroplasmic ratio of the A3243G mitochondrial DNA mutation in single pancreatic beta cells.

Authors:  S Lynn; G M Borthwick; R M Charnley; M Walker; D M Turnbull
Journal:  Diabetologia       Date:  2003-01-31       Impact factor: 10.122

Review 2.  Mitochondrial threshold effects.

Authors:  Rodrigue Rossignol; Benjamin Faustin; Christophe Rocher; Monique Malgat; Jean-Pierre Mazat; Thierry Letellier
Journal:  Biochem J       Date:  2003-03-15       Impact factor: 3.857

3.  A history of mitochondrial diseases.

Authors:  Salvatore Dimauro
Journal:  J Inherit Metab Dis       Date:  2010-05-21       Impact factor: 4.982

Review 4.  The clinical maze of mitochondrial neurology.

Authors:  Salvatore DiMauro; Eric A Schon; Valerio Carelli; Michio Hirano
Journal:  Nat Rev Neurol       Date:  2013-07-09       Impact factor: 42.937

5.  Random genetic drift determines the level of mutant mtDNA in human primary oocytes.

Authors:  D T Brown; D C Samuels; E M Michael; D M Turnbull; P F Chinnery
Journal:  Am J Hum Genet       Date:  2000-12-29       Impact factor: 11.025

6.  Mitochondrial dysfunction and mitochondrial DNA mutations in atherosclerotic complications in diabetes.

Authors:  Dimitry A Chistiakov; Igor A Sobenin; Yuri V Bobryshev; Alexander N Orekhov
Journal:  World J Cardiol       Date:  2012-05-26

7.  A novel mutation in the mitochondrial tRNA(Pro) gene associated with late-onset ataxia, retinitis pigmentosa, deafness, leukoencephalopathy and complex I deficiency.

Authors:  Paola Da Pozzo; Elena Cardaioli; Edoardo Malfatti; Gian Nicola Gallus; Alessandro Malandrini; Carmen Gaudiano; Gianna Berti; Federica Invernizzi; Massimo Zeviani; Antonio Federico
Journal:  Eur J Hum Genet       Date:  2009-02-18       Impact factor: 4.246

8.  Preimplantation genetic diagnosis for mitochondrial DNA disorders: ethical guidance for clinical practice.

Authors:  Annelien Bredenoord; Wybo Dondorp; Guido Pennings; Christine de Die-Smulders; Bert Smeets; Guido de Wert
Journal:  Eur J Hum Genet       Date:  2009-05-27       Impact factor: 4.246

Review 9.  Mitochondrial disease: mutations and mechanisms.

Authors:  Matthew McKenzie; Danae Liolitsa; Michael G Hanna
Journal:  Neurochem Res       Date:  2004-03       Impact factor: 3.996

10.  Neonatal presentation of ventricular tachycardia and a Reye-like syndrome episode associated with disturbed mitochondrial energy metabolism.

Authors:  Fernando Scaglia; Angela E Scheuerle; Jeffrey A Towbin; Dawna L Armstrong; Lawrence Sweetman; Lee-Jun C Wong
Journal:  BMC Pediatr       Date:  2002-12-30       Impact factor: 2.125

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