Literature DB >> 1649912

Multiple defects of the mitochondrial respiratory chain in a mitochondrial encephalopathy (MERRF): a clinical, biochemical and molecular study.

L A Bindoff1, C Desnuelle, M A Birch-Machin, J F Pellissier, G Serratrice, C Dravet, M Bureau, N Howell, D M Turnbull.   

Abstract

We describe a young man with a progressive neurological disorder including myoclonus, mental retardation, muscle weakness and a mitochondrial myopathy (myoclonus epilepsy and ragged red fibres--MERRF). Multiple abnormalities of the mitochondrial respiratory chain in skeletal muscle are shown by direct measurement of the flux through the individual complexes, low-temperature redox spectroscopy and decreased immunodetectable subunits of complexes I and IV by immunoblotting. No abnormality of mitochondrial DNA was found. This is the first report of combined defects of complexes I, III and IV as a cause of this clinical syndrome. However, we propose that the occurrence of multiple respiratory chain defects may be more common than previously recognised and that this particular combination of defects, involving complexes I, III and IV, may be the predominant biochemical abnormality in MERRF.

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Year:  1991        PMID: 1649912     DOI: 10.1016/0022-510x(91)90088-o

Source DB:  PubMed          Journal:  J Neurol Sci        ISSN: 0022-510X            Impact factor:   3.181


  12 in total

Review 1.  Molecular biology of neurological diseases.

Authors:  W J Cumming
Journal:  Postgrad Med J       Date:  1992-04       Impact factor: 2.401

2.  Potentially diagnostic electron paramagnetic resonance spectra elucidate the underlying mechanism of mitochondrial dysfunction in the deoxyguanosine kinase deficient rat model of a genetic mitochondrial DNA depletion syndrome.

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3.  Intracellular mitochondrial triplasmy in a patient with two heteroplasmic base changes.

Authors:  S K Bidooki; M A Johnson; Z Chrzanowska-Lightowlers; L A Bindoff; R N Lightowlers
Journal:  Am J Hum Genet       Date:  1997-06       Impact factor: 11.025

4.  Human equilibrative nucleoside transporter-3 (hENT3) spectrum disorder mutations impair nucleoside transport, protein localization, and stability.

Authors:  Nayoung Kang; Ah Hyun Jun; Yangzom Doma Bhutia; Natarajan Kannan; Jashvant D Unadkat; Rajgopal Govindarajan
Journal:  J Biol Chem       Date:  2010-07-01       Impact factor: 5.157

5.  Parkin-dependent degradation of the F-box protein Fbw7β promotes neuronal survival in response to oxidative stress by stabilizing Mcl-1.

Authors:  Susanna Ekholm-Reed; Matthew S Goldberg; Michael G Schlossmacher; Steven I Reed
Journal:  Mol Cell Biol       Date:  2013-07-15       Impact factor: 4.272

6.  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

7.  Distribution and threshold expression of the tRNA(Lys) mutation in skeletal muscle of patients with myoclonic epilepsy and ragged-red fibers (MERRF).

Authors:  L Boulet; G Karpati; E A Shoubridge
Journal:  Am J Hum Genet       Date:  1992-12       Impact factor: 11.025

8.  A topoisomerase II cleavage site is associated with a novel mitochondrial DNA deletion.

Authors:  R B Blok; D R Thorburn; G N Thompson; H H Dahl
Journal:  Hum Genet       Date:  1995-01       Impact factor: 4.132

9.  A novel mtDNA deletion in an infant with Pearson syndrome.

Authors:  R Kapsa; G N Thompson; D R Thorburn; H H Dahl; S Marzuki; E Byrne; R B Blok
Journal:  J Inherit Metab Dis       Date:  1994       Impact factor: 4.982

Review 10.  The in-depth evaluation of suspected mitochondrial disease.

Authors:  Richard H Haas; Sumit Parikh; Marni J Falk; Russell P Saneto; Nicole I Wolf; Niklas Darin; Lee-Jun Wong; Bruce H Cohen; Robert K Naviaux
Journal:  Mol Genet Metab       Date:  2008-02-01       Impact factor: 4.797

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