Literature DB >> 9700597

mtDNA depletion and impairment of mitochondrial function in a case of a multisystem disorder including severe myopathy.

E J Kirches1, K Winkler, M Warich-Kirches, R Szibor, F Wien, W S Kunz, P von Bossanyi, P K Bajaj, K Dietzmann.   

Abstract

The ratio of mtDNA and a nuclear reference gene was estimated by Southern blotting in the skeletal muscle DNA of a 3-year-old girl who suffered from congenital brain damage, focal epilepsy, hepatomegaly, malabsorption syndrome and severe myopathy. The signal ratio of mtDNA versus 18S rDNA was 22% of the mean value obtained from controls. No major deletions or insertions were found and the MERRF, MELAS and NARP mutations were ruled out. Mitochondrial DNA-encoded enzyme activities and mitochondrial respiration were reduced. The analysis of the NAD(P)H and flavoprotein redox states of intact fibres revealed the presence of mitochondrial dysfunction. In tissue sections a moderate elevation of type I and type II fibre diameter variation was detected, aberrant NADH- and succinate dehydrogenase staining and some ragged red fibres. This suggested that a mitochondrial disorder caused by a decrease in the amount of intact wild-type mtDNA was responsible for the severe myopathy.

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Year:  1998        PMID: 9700597     DOI: 10.1023/a:1005306725644

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  23 in total

1.  A MERRF/PEO overlap syndrome associated with the mitochondrial DNA 3243 mutation.

Authors:  A Verma; C T Moraes; R T Shebert; W G Bradley
Journal:  Neurology       Date:  1996-05       Impact factor: 9.910

2.  Enzymatic activities of mitochondrial respiratory complexes from children muscular biopsies. Age-related evolutions.

Authors:  E Lefai; A Terrier-Cayre; A Vincent; O Boespflug-Tanguy; A Tanguy; S Alziari
Journal:  Biochim Biophys Acta       Date:  1995-02-14

3.  Sequence and organization of the human mitochondrial genome.

Authors:  S Anderson; A T Bankier; B G Barrell; M H de Bruijn; A R Coulson; J Drouin; I C Eperon; D P Nierlich; B A Roe; F Sanger; P H Schreier; A J Smith; R Staden; I G Young
Journal:  Nature       Date:  1981-04-09       Impact factor: 49.962

4.  Polyneuropathy, ophthalmoplegia, leukoencephalopathy, and intestinal pseudo-obstruction: POLIP syndrome.

Authors:  L T Simon; D S Horoupian; L J Dorfman; M Marks; M K Herrick; P Wasserstein; M E Smith
Journal:  Ann Neurol       Date:  1990-09       Impact factor: 10.422

5.  Low levels of mitochondrial transcription factor A in mitochondrial DNA depletion.

Authors:  N G Larsson; A Oldfors; E Holme; D A Clayton
Journal:  Biochem Biophys Res Commun       Date:  1994-05-16       Impact factor: 3.575

6.  Biochemical evidence for nuclear gene involvement in phenotype of non-syndromic deafness associated with mitochondrial 12S rRNA mutation.

Authors:  M X Guan; N Fischel-Ghodsian; G Attardi
Journal:  Hum Mol Genet       Date:  1996-07       Impact factor: 6.150

7.  Early-onset fatal encephalomyopathy associated with severe mtDNA depletion.

Authors:  V Paquis-Flucklinger; J F Pellissier; J Camboulives; B Chabrol; A Saunières; M F Monfort; H Giudicelli; C Desnuelle
Journal:  Eur J Pediatr       Date:  1995-07       Impact factor: 3.183

8.  Insulin resistance associated with maternally inherited diabetes and deafness.

Authors:  S S Gebhart; J M Shoffner; D Koontz; A Kaufman; D Wallace
Journal:  Metabolism       Date:  1996-04       Impact factor: 8.694

Review 9.  Mitochondrial DNA mutations in diseases of energy metabolism.

Authors:  D C Wallace
Journal:  J Bioenerg Biomembr       Date:  1994-06       Impact factor: 2.945

10.  Variation in mitochondrial DNA levels in muscle from normal controls. Is depletion of mtDNA in patients with mitochondrial myopathy a distinct clinical syndrome.

Authors:  J Poulton; C Sewry; C G Potter; T Bougeron; D Chretien; F A Wijburg; K J Morten; G Brown
Journal:  J Inherit Metab Dis       Date:  1995       Impact factor: 4.982

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