Literature DB >> 8254046

Two novel pathogenic mitochondrial DNA mutations affecting organelle number and protein synthesis. Is the tRNA(Leu(UUR)) gene an etiologic hot spot?

C T Moraes1, F Ciacci, E Bonilla, C Jansen, M Hirano, N Rao, R E Lovelace, L P Rowland, E A Schon, S DiMauro.   

Abstract

We identified two patients with pathogenic single nucleotide changes in two different mitochondrial tRNA genes: the first mutation in the tRNA(Asn) gene, and the ninth known mutation in the tRNA(Leu(UUR)) gene. The mutation in tRNA(Asn) was associated with isolated ophthalmoplegia, whereas the mutation in tRNA(Leu(UUR)) caused a neurological syndrome resembling MERRF (myoclonus epilepsy and ragged-red fibers) plus optic neuropathy, retinopathy, and diabetes. Both mutations were heteroplasmic, with higher percentages of mutant mtDNA in affected tissues, and undetectable levels in maternal relatives. Analysis of single muscle fibers indicated that morphological and biochemical alterations appeared only when the proportions of mutant mtDNA exceeded 90% of the total cellular mtDNA pool. The high incidence of mutations in the tRNA(Leu(UUR)) gene suggests that this region is an "etiologic hot spot" in mitochondrial disease.

Entities:  

Mesh:

Substances:

Year:  1993        PMID: 8254046      PMCID: PMC288494          DOI: 10.1172/JCI116913

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  52 in total

1.  Cytochrome c oxidase deficiency in Leigh syndrome.

Authors:  S DiMauro; S Servidei; M Zeviani; M DiRocco; D C DeVivo; S DiDonato; G Uziel; K Berry; G Hoganson; S D Johnsen
Journal:  Ann Neurol       Date:  1987-10       Impact factor: 10.422

2.  A new mitochondrial disease associated with mitochondrial DNA heteroplasmy.

Authors:  I J Holt; A E Harding; R K Petty; J A Morgan-Hughes
Journal:  Am J Hum Genet       Date:  1990-03       Impact factor: 11.025

3.  Mitochondrial myopathies: clinical and biochemical features of 30 patients with major deletions of muscle mitochondrial DNA.

Authors:  I J Holt; A E Harding; J M Cooper; A H Schapira; A Toscano; J B Clark; J A Morgan-Hughes
Journal:  Ann Neurol       Date:  1989-12       Impact factor: 10.422

4.  Compilation of tRNA sequences and sequences of tRNA genes.

Authors:  M Sprinzl; T Hartmann; J Weber; J Blank; R Zeidler
Journal:  Nucleic Acids Res       Date:  1989       Impact factor: 16.971

Review 5.  Biogenesis of mitochondria.

Authors:  G Attardi; G Schatz
Journal:  Annu Rev Cell Biol       Date:  1988

6.  Termination of transcription in human mitochondria: identification and purification of a DNA binding protein factor that promotes termination.

Authors:  B Kruse; N Narasimhan; G Attardi
Journal:  Cell       Date:  1989-07-28       Impact factor: 41.582

7.  Transcription and translation of deleted mitochondrial genomes in Kearns-Sayre syndrome: implications for pathogenesis.

Authors:  H Nakase; C T Moraes; R Rizzuto; A Lombes; S DiMauro; E A Schon
Journal:  Am J Hum Genet       Date:  1990-03       Impact factor: 11.025

8.  Mitochondrial DNA deletions in progressive external ophthalmoplegia and Kearns-Sayre syndrome.

Authors:  C T Moraes; S DiMauro; M Zeviani; A Lombes; S Shanske; A F Miranda; H Nakase; E Bonilla; L C Werneck; S Servidei
Journal:  N Engl J Med       Date:  1989-05-18       Impact factor: 91.245

9.  The mitochondrial tRNA(Leu)(UUR)) mutation in MELAS: a model for pathogenesis.

Authors:  E A Schon; Y Koga; M Davidson; C T Moraes; M P King
Journal:  Biochim Biophys Acta       Date:  1992-07-17

10.  Geographic variation in human mitochondrial DNA from Papua New Guinea.

Authors:  M Stoneking; L B Jorde; K Bhatia; A C Wilson
Journal:  Genetics       Date:  1990-03       Impact factor: 4.562

View more
  42 in total

Review 1.  Clinical mitochondrial genetics.

Authors:  P F Chinnery; N Howell; R M Andrews; D M Turnbull
Journal:  J Med Genet       Date:  1999-06       Impact factor: 6.318

Review 2.  Progressive myoclonic epilepsies: review of clinical, molecular and therapeutic aspects.

Authors:  Luis Felipe Mendonça de Siqueira
Journal:  J Neurol       Date:  2010-07-01       Impact factor: 4.849

3.  Sequence variation in the tRNA genes of human mitochondrial DNA.

Authors:  Tiina Vilmi; Jukka S Moilanen; Saara Finnilä; Kari Majamaa
Journal:  J Mol Evol       Date:  2005-05       Impact factor: 2.395

4.  Maternally inherited cardiomyopathy and hearing loss associated with a novel mutation in the mitochondrial tRNA(Lys) gene (G8363A).

Authors:  F M Santorelli; S C Mak; M El-Schahawi; C Casali; S Shanske; T Z Baram; R E Madrid; S DiMauro
Journal:  Am J Hum Genet       Date:  1996-05       Impact factor: 11.025

5.  Evidence that specific mtDNA point mutations may not accumulate in skeletal muscle during normal human aging.

Authors:  F Pallotti; X Chen; E Bonilla; E A Schon
Journal:  Am J Hum Genet       Date:  1996-09       Impact factor: 11.025

6.  Comprehensive, rapid and sensitive detection of sequence variants of human mitochondrial tRNA genes.

Authors:  Y Michikawa; G Hofhaus; L S Lerman; G Attardi
Journal:  Nucleic Acids Res       Date:  1997-06-15       Impact factor: 16.971

Review 7.  Mitochondrial DNA mutations and pathogenesis.

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

8.  The expression level of the voltage-dependent anion channel controls life and death of the cell.

Authors:  Salah Abu-Hamad; Sara Sivan; Varda Shoshan-Barmatz
Journal:  Proc Natl Acad Sci U S A       Date:  2006-04-03       Impact factor: 11.205

9.  Point mutation of the mitochondrial tRNA(Leu) gene (A 3243 G) in maternally inherited hypertrophic cardiomyopathy, diabetes mellitus, renal failure, and sensorineural deafness.

Authors:  S Manouvrier; A Rötig; G Hannebique; J D Gheerbrandt; G Royer-Legrain; A Munnich; M Parent; J P Grünfeld; C Largilliere; A Lombes
Journal:  J Med Genet       Date:  1995-08       Impact factor: 6.318

Review 10.  [Mitochondrial hearing impairment. Background, genetic predisposition and possibilities for diagnosis].

Authors:  K Riemann; M Pfister; N Blin; S Kupka
Journal:  HNO       Date:  2004-06       Impact factor: 1.284

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.