Literature DB >> 2852217

Progression from MERRF to MELAS phenotype in a patient with combined respiratory complex I and IV deficiencies.

E Byrne1, I Trounce, X Dennett, B Gilligan, J B Morley, S Marzuki.   

Abstract

Identical twins developed myoclonic epilepsy in their teens. One twin remained mildly affected but the other went on to develop sensorineural deafness and ataxia with lactic acidosis and ragged red fibres leading to a diagnosis of mitochondrial encephalopathy. Multiple stroke-like episodes with hemiparesis followed, indicating progression from a MERRF to a MELAS phenotype. Biochemical studies revealed a severe deficiency of mitochondrial NADH-ubiquinone reductase and a moderate deficiency of cytochrome aa3. Western immunoblotting experiments using polyclonal antibodies raised against human placental cytochrome oxidase identified a similar profile of bands to those seen in controls, supporting the view that cytochrome aa3 deficiency in this case may be a secondary consequence of a failure of assembly related to a severe proximal respiratory chain defect.

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Year:  1988        PMID: 2852217     DOI: 10.1016/0022-510x(88)90229-8

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


  6 in total

1.  Fatal cerebral hemorrhage in mitochondrial encephalomyopathy. Clinical and pathological data of a case.

Authors:  Haruhisa Kato; Masanobu Uchigata; Mutsumi Iijima; Seiichiro Shimizu; Ikuya Nonaka; Yuichi Goto
Journal:  J Neurol       Date:  2005-11-14       Impact factor: 4.849

2.  Normal variants of human mitochondrial DNA and translation products: the building of a reference data base.

Authors:  S Marzuki; A S Noer; P Lertrit; D Thyagarajan; R Kapsa; P Utthanaphol; E Byrne
Journal:  Hum Genet       Date:  1991-12       Impact factor: 4.132

3.  Clinical and laboratory findings in referrals for mitochondrial DNA analysis.

Authors:  P J Lamont; R Surtees; C E Woodward; J V Leonard; N W Wood; A E Harding
Journal:  Arch Dis Child       Date:  1998-07       Impact factor: 3.791

4.  Cytochrome c oxidase activity is deficient in blood vessels of patients with myoclonus epilepsy with ragged-red fibers.

Authors:  H Hasegawa; T Matsuoka; Y Goto; I Nonaka
Journal:  Acta Neuropathol       Date:  1993       Impact factor: 17.088

5.  Functional respiratory chain studies in mitochondrial cytopathies. Support for mitochondrial DNA heteroplasmy in myoclonus epilepsy and ragged red fibers (MERRF) syndrome.

Authors:  E Byrne; I Trounce; S Marzuki; X Dennett; S F Berkovic; S Davis; M Tanaka; T Ozawa
Journal:  Acta Neuropathol       Date:  1991       Impact factor: 17.088

6.  Epilepsy in adults with mitochondrial disease: A cohort study.

Authors:  Roger G Whittaker; Helen E Devine; Grainne S Gorman; Andrew M Schaefer; Rita Horvath; Yi Ng; Victoria Nesbitt; Nichola Z Lax; Robert McFarland; Mark O Cunningham; Robert W Taylor; Douglass M Turnbull
Journal:  Ann Neurol       Date:  2015-11-17       Impact factor: 10.422

  6 in total

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