Literature DB >> 15657614

LHON/MELAS overlap syndrome associated with a mitochondrial MTND1 gene mutation.

Emma L Blakely1, Rajith de Silva, Andrew King, Verena Schwarzer, Tim Harrower, Gervase Dawidek, Douglass M Turnbull, Robert W Taylor.   

Abstract

Pathogenic point mutations in the mitochondrial MTND1 gene have previously been described in association with two distinct clinical phenotypes -- Leber hereditary optic neuropathy (LHON) and mitochondrial myopathy, encephalopathy, lactic acidosis and stroke-like episodes (MELAS). Here we report the first heteroplasmic mitochondrial DNA (mtDNA) point mutation (3376G>A) in the MTND1 gene associated with an overlap syndrome comprising the clinical features of both LHON and MELAS. Muscle histochemistry revealed subtle mitochondrial abnormalities, while biochemical analysis showed an isolated complex I deficiency. Our findings serve to highlight the growing importance of mutations in mitochondrial complex I structural genes in MELAS and its associated overlap syndromes.

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Year:  2005        PMID: 15657614     DOI: 10.1038/sj.ejhg.5201363

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   4.246


  21 in total

1.  [Leber's hereditary optic neuropathy].

Authors:  B Leo-Kottler; B Wissinger
Journal:  Ophthalmologe       Date:  2011-12       Impact factor: 1.059

Review 2.  Mitochondrial complex I: structure, function and pathology.

Authors:  Rolf J R J Janssen; Leo G Nijtmans; Lambert P van den Heuvel; Jan A M Smeitink
Journal:  J Inherit Metab Dis       Date:  2006-07-11       Impact factor: 4.982

3.  Critical roles of subunit NuoH (ND1) in the assembly of peripheral subunits with the membrane domain of Escherichia coli NDH-1.

Authors:  Prem Kumar Sinha; Jesus Torres-Bacete; Eiko Nakamaru-Ogiso; Norma Castro-Guerrero; Akemi Matsuno-Yagi; Takao Yagi
Journal:  J Biol Chem       Date:  2009-02-03       Impact factor: 5.157

Review 4.  The neuro-ophthalmology of mitochondrial disease.

Authors:  J Alexander Fraser; Valérie Biousse; Nancy J Newman
Journal:  Surv Ophthalmol       Date:  2010-05-14       Impact factor: 6.048

Review 5.  Mitochondrial optic neuropathies - disease mechanisms and therapeutic strategies.

Authors:  Patrick Yu-Wai-Man; Philip G Griffiths; Patrick F Chinnery
Journal:  Prog Retin Eye Res       Date:  2010-11-26       Impact factor: 21.198

6.  Mitochondrial Respiratory Defect Causes Dysfunctional Lactate Turnover via AMP-activated Protein Kinase Activation in Human-induced Pluripotent Stem Cell-derived Hepatocytes.

Authors:  Ilkyun Im; Mi-Jin Jang; Seung Ju Park; Sang-Hee Lee; Jin-Ho Choi; Han-Wook Yoo; Seyun Kim; Yong-Mahn Han
Journal:  J Biol Chem       Date:  2015-10-21       Impact factor: 5.157

7.  Clinical approach to optic neuropathies.

Authors:  Raed Behbehani
Journal:  Clin Ophthalmol       Date:  2007-09

8.  Mutation analysis of Leber's hereditary optic neuropathy using a multi-gene panel.

Authors:  Yu Dai; Chenghui Wang; Zhipeng Nie; Jiamin Han; Ting Chen; Xiaoxu Zhao; Cheng Ai; Yanchun Ji; Tao Gao; Pingping Jiang
Journal:  Biomed Rep       Date:  2017-11-08

9.  The 13042G --> A/ND5 mutation in mtDNA is pathogenic and can be associated also with a prevalent ocular phenotype.

Authors:  M L Valentino; P Barboni; C Rengo; A Achilli; A Torroni; R Lodi; C Tonon; B Barbiroli; F Fortuna; P Montagna; A Baruzzi; V Carelli
Journal:  J Med Genet       Date:  2006-07       Impact factor: 6.318

Review 10.  The optic nerve: a "mito-window" on mitochondrial neurodegeneration.

Authors:  Alessandra Maresca; Chiara la Morgia; Leonardo Caporali; Maria Lucia Valentino; Valerio Carelli
Journal:  Mol Cell Neurosci       Date:  2012-08-15       Impact factor: 4.314

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