Literature DB >> 2604380

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

I J Holt1, A E Harding, J M Cooper, A H Schapira, A Toscano, J B Clark, J A Morgan-Hughes.   

Abstract

Analysis of mitochondrial DNA (mtDNA) in muscle and blood from 72 patients with mitochondrial myopathy showed that 30 had major deletions of a variable proportion of muscle mtDNA. All of these 30 patients presented with progressive external ophthalmoplegia and limb weakness, and 8 had the additional features of the Kearns-Sayre syndrome. Of the 42 patients without detectable muscle mtDNA deletions, 10 had progressive external ophthalmoplegia and limb weakness, 2 had the Kearns-Sayre syndrome, 11 had limb weakness without extraocular involvement, and 19 had multisystem disorders predominantly affecting the central nervous system. Only 2 patients with mtDNA deletions had clinically affected relatives, compared with 10 of those without deletions. In the 4 patients with polarographic defects exclusively involving complex I (NADH coenzyme Q reductase), the deleted protein-coding genes were confined to those for complex I subunits. Thirteen other patients with apparently identical deletions had variable clinical and biochemical features. Immunoblots of complex I polypeptides from patients with deletions were either indistinguishable from controls or showed only a mild generalized decrease in all identifiable subunits.

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Year:  1989        PMID: 2604380     DOI: 10.1002/ana.410260603

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


  57 in total

1.  Detection of extremely low levels of wild-type mitochondrial DNA in the liver of a patient with Pearson syndrome by a sensitive PCR assay.

Authors:  D D de Vries; W Ruitenbeek; B A van Oost
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

2.  The mutant mitochondrial genes in mitochondrial myopathy, encephalopathy, lactic acidosis and stroke-like episodes (MELAS) were selectively amplified through generations.

Authors:  Y Kobayashi; K Ichihashi; S Ohta; K Nihei; Y Kagawa; M Yanagisawa; M Y Momoi
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

3.  Prenatal diagnosis of mitochondrial DNA8993 T----G disease.

Authors:  A E Harding; I J Holt; M G Sweeney; M Brockington; M B Davis
Journal:  Am J Hum Genet       Date:  1992-03       Impact factor: 11.025

Review 4.  Deletions of the mitochondrial genome.

Authors:  A E Harding; S R Hammans
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

5.  The other genome.

Authors:  A E Harding
Journal:  BMJ       Date:  1991-08-17

6.  Mitochondrial genome: defects, disease, and evolution.

Authors:  A Clarke
Journal:  J Med Genet       Date:  1990-07       Impact factor: 6.318

7.  Pearson syndrome and mitochondrial encephalomyopathy in a patient with a deletion of mtDNA.

Authors:  M A McShane; S R Hammans; M Sweeney; I J Holt; T J Beattie; E M Brett; A E Harding
Journal:  Am J Hum Genet       Date:  1991-01       Impact factor: 11.025

8.  Orbital magnetic resonance imaging of extraocular muscles in chronic progressive external ophthalmoplegia: specific diagnostic findings.

Authors:  Maria Carolina Ortube; Rahul Bhola; Joseph L Demer
Journal:  J AAPOS       Date:  2006-10       Impact factor: 1.220

9.  Missense mutations in the beta-myosin heavy-chain gene cause central core disease in hypertrophic cardiomyopathy.

Authors:  L Fananapazir; M C Dalakas; F Cyran; G Cohn; N D Epstein
Journal:  Proc Natl Acad Sci U S A       Date:  1993-05-01       Impact factor: 11.205

10.  Myopathy in vitamin E deficient rats: muscle fibre necrosis associated with disturbances of mitochondrial function.

Authors:  P K Thomas; J M Cooper; R H King; J M Workman; A H Schapira; M A Goss-Sampson; D P Muller
Journal:  J Anat       Date:  1993-12       Impact factor: 2.610

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