Literature DB >> 8164833

Mitochondrial neurogastrointestinal encephalomyopathy (MNGIE): clinical, biochemical, and genetic features of an autosomal recessive mitochondrial disorder.

M Hirano1, G Silvestri, D M Blake, A Lombes, C Minetti, E Bonilla, A P Hays, R E Lovelace, I Butler, T E Bertorini.   

Abstract

We studied the clinical, biochemical, and genetic features of eight patients with the autosomal recessive mitochondrial syndrome mitochondrial neurogastrointestinal encephalomyopathy (MNGIE). MNGIE is clinically characterized by ophthalmoparesis, peripheral neuropathy, leukoencephalopathy, gastrointestinal symptoms (recurrent nausea, vomiting, or diarrhea) with intestinal dysmotility, and histologically abnormal mitochondria in muscle. Brain MRI scans were consistent with leukodystrophy in seven patients examined. Nerve conduction and EMG studies were compatible with a sensorimotor neuropathy; quantitative EMG of two patients suggested a myogenic process. Muscle mitochondrial enzyme analysis revealed a partial defect of cytochrome c oxidase activity in five patients; three had additional respiratory chain enzyme defects. Two patients had isolated complex I defects, and one had normal respiratory chain function. Southern blot analysis revealed multiple deletions of mitochondrial DNA in four of eight patients.

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Year:  1994        PMID: 8164833     DOI: 10.1212/wnl.44.4.721

Source DB:  PubMed          Journal:  Neurology        ISSN: 0028-3878            Impact factor:   9.910


  83 in total

Review 1.  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

2.  Clinical and genetic spectrum of mitochondrial neurogastrointestinal encephalomyopathy.

Authors:  Caterina Garone; Saba Tadesse; Michio Hirano
Journal:  Brain       Date:  2011-09-20       Impact factor: 13.501

3.  Substoichiometric shifting in the plant mitochondrial genome is influenced by a gene homologous to MutS.

Authors:  Ricardo V Abdelnoor; Ryan Yule; Annakaisa Elo; Alan C Christensen; Gilbert Meyer-Gauen; Sally A Mackenzie
Journal:  Proc Natl Acad Sci U S A       Date:  2003-05-01       Impact factor: 11.205

4.  Twinkle and POLG defects enhance age-dependent accumulation of mutations in the control region of mtDNA.

Authors:  Sjoerd Wanrooij; Petri Luoma; Gert van Goethem; Christine van Broeckhoven; Anu Suomalainen; Johannes N Spelbrink
Journal:  Nucleic Acids Res       Date:  2004-06-04       Impact factor: 16.971

5.  Clinical and biochemical improvement following HSCT in a patient with MNGIE: 1-year follow-up.

Authors:  F Sicurelli; M A Carluccio; F Toraldo; M Tozzi; A Bucalossi; M Lenoci; G Jacomelli; V Micheli; E Cardaioli; M Mondelli; A Federico; G Marotta; M T Dotti
Journal:  J Neurol       Date:  2012-04-25       Impact factor: 4.849

Review 6.  CoQ(10) deficiencies and MNGIE: two treatable mitochondrial disorders.

Authors:  Michio Hirano; Caterina Garone; Catarina M Quinzii
Journal:  Biochim Biophys Acta       Date:  2012-01-18

Review 7.  Mitochondrial DNA depletion syndromes: review and updates of genetic basis, manifestations, and therapeutic options.

Authors:  Ayman W El-Hattab; Fernando Scaglia
Journal:  Neurotherapeutics       Date:  2013-04       Impact factor: 7.620

8.  Clinicopathological aspects of the neuropathy of neurogastrointestinal encephalomyopathy (MNGIE) in four patients including two with a Charcot-Marie-Tooth presentation.

Authors:  Gérard Said; Catherine Lacroix; Violaine Planté-Bordeneuve; Bernard Messing; Abdelhamid Slama; Pascal Crenn; Annie Nivelon-Chevallier; Laurent Bedenne; Pierre Soichot; E Manceau; Daniel Rigaud; Anne Guiochon-Mantel; Claude Matuchansky
Journal:  J Neurol       Date:  2005-03-07       Impact factor: 4.849

Review 9.  Progressive external ophthalmoplegia characterized by multiple deletions of mitochondrial DNA: unraveling the pathogenesis of human mitochondrial DNA instability and the initiation of a genetic classification.

Authors:  Gert Van Goethem; Jean-Jacques Martin; Christine Van Broeckhoven
Journal:  Neuromolecular Med       Date:  2003       Impact factor: 3.843

10.  Encephalomyopathies caused by abnormal nuclear-mitochondrial intergenomic cross-talk.

Authors:  C Lamperti; M Zeviani
Journal:  Acta Myol       Date:  2009-07
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