Literature DB >> 8250532

The mutation at nt 8993 of mitochondrial DNA is a common cause of Leigh's syndrome.

F M Santorelli1, S Shanske, A Macaya, D C DeVivo, S DiMauro.   

Abstract

Twelve patients with Leigh's syndrome from 10 families harbored a T > G point mutation at nt 8993 of mtDNA. This mutation, initially associated with neurogenic weakness, ataxia, and retinitis pigmentosa, was later found to result in the Leigh phenotype when present in a high percentage. In our patients, the mutation was heteroplasmic, maternally inherited, and appeared to segregate rapidly within the pedigrees. Quantitative analysis revealed a good correlation between percentage of mutant mitochondrial genomes and severity of the clinical phenotype. The mutation was not found in > 200 patients with other mitochondrial encephalomyopathies or in controls. Mitochondrial enzyme activities were normal in all but 1 patient, and there were no ragged-red fibers in the muscle biopsy. Lactic acidosis was present in 92% of patients. Our findings suggest that the mtDNA nt 8993 mutation is a relatively common cause of Leigh's syndrome.

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Year:  1993        PMID: 8250532     DOI: 10.1002/ana.410340612

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


  51 in total

1.  Genetic counseling and prenatal diagnosis for the mitochondrial DNA mutations at nucleotide 8993.

Authors:  S L White; V R Collins; R Wolfe; M A Cleary; S Shanske; S DiMauro; H H Dahl; D R Thorburn
Journal:  Am J Hum Genet       Date:  1999-08       Impact factor: 11.025

2.  Mitochondrial DNA mutations at nucleotide 8993 show a lack of tissue- or age-related variation.

Authors:  S L White; S Shanske; J J McGill; H Mountain; M T Geraghty; S DiMauro; H H Dahl; D R Thorburn
Journal:  J Inherit Metab Dis       Date:  1999-12       Impact factor: 4.982

Review 3.  Disorders related to mitochondrial membranes: pathology of the respiratory chain and neurodegeneration.

Authors:  S Di Donato
Journal:  J Inherit Metab Dis       Date:  2000-05       Impact factor: 4.982

Review 4.  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

5.  Leigh syndrome caused by mitochondrial DNA G13513A mutation: frequency and clinical features in Japan.

Authors:  Akira Sudo; Shiho Honzawa; Ikuya Nonaka; Yu-Ichi Goto
Journal:  J Hum Genet       Date:  2004-01-17       Impact factor: 3.172

Review 6.  Convergent mechanisms in etiologically-diverse dystonias.

Authors:  Valerie B Thompson; H A Jinnah; Ellen J Hess
Journal:  Expert Opin Ther Targets       Date:  2011-12-03       Impact factor: 6.902

7.  Mitochondrial DNA 11777C>A mutation associated Leigh syndrome: case report with a review of the previously described pedigrees.

Authors:  Kinga Hadzsiev; Anita Maasz; Peter Kisfali; Endre Kalman; Eva Gomori; Endre Pal; Ervin Berenyi; Katalin Komlosi; Bela Melegh
Journal:  Neuromolecular Med       Date:  2010-05-26       Impact factor: 3.843

8.  The development of novel quantification assay for mitochondrial DNA heteroplasmy aimed at preimplantation genetic diagnosis of Leigh encephalopathy.

Authors:  Hiroto Tajima; Kou Sueoka; Sung Yung Moon; Akira Nakabayashi; Tomoyoshi Sakurai; Yukitaka Murakoshi; Hiroyoshi Watanabe; Soukichi Iwata; Tsuyoshi Hashiba; Shingo Kato; Yu-Ichi Goto; Yasunori Yoshimura
Journal:  J Assist Reprod Genet       Date:  2007-03-08       Impact factor: 3.412

9.  Multiple presentation of mitochondrial disorders.

Authors:  A Nissenkorn; A Zeharia; D Lev; A Fatal-Valevski; V Barash; A Gutman; S Harel; T Lerman-Sagie
Journal:  Arch Dis Child       Date:  1999-09       Impact factor: 3.791

10.  New insights into the bioenergetics of mitochondrial disorders using intracellular ATP reporters.

Authors:  Carl D Gajewski; Lichuan Yang; Eric A Schon; Giovanni Manfredi
Journal:  Mol Biol Cell       Date:  2003-06-27       Impact factor: 4.138

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