Literature DB >> 15054399

A novel point mutation in the mitochondrial tRNA(Trp) gene produces a neurogastrointestinal syndrome.

Katharina Maniura-Weber1, Robert W Taylor, Margaret A Johnson, Zofia Chrzanowska-Lightowlers, Andrew A M Morris, Charles P J Charlton, Douglass M Turnbull, Laurence A Bindoff.   

Abstract

We report a novel, heteroplasmic point mutation in the mitochondrial tRNA for tryptophan at position 5532. The mutation was present in all the tissues studied and segregated with the biochemical defect, with higher levels of mutation present in cytochrome c oxidase-deficient muscle fibres. The patient manifested a neurogastrointestinal syndrome with features including failure to thrive, psychomotor retardation, ophthalmoplegia, sensorineural deafness and encephalopathy together with vomiting, diarrhoea and colitis.

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Year:  2004        PMID: 15054399     DOI: 10.1038/sj.ejhg.5201185

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


  11 in total

Review 1.  Multisystem manifestations of mitochondrial disorders.

Authors:  Stefano Di Donato
Journal:  J Neurol       Date:  2009-03-01       Impact factor: 4.849

2.  Mitochondrial myopathy associated with a novel 5522G>A mutation in the mitochondrial tRNA(Trp) gene.

Authors:  Ivo Barić; Ksenija Fumić; Danijela Petković Ramadža; Wolfgang Sperl; Franz A Zimmermann; Diana Muačević-Katanec; Zoran Mitrović; Leo Pažanin; Ljerka Cvitanović Šojat; Tihomir Kekez; Zeljko Reiner; Johannes A Mayr
Journal:  Eur J Hum Genet       Date:  2012-12-12       Impact factor: 4.246

3.  The tRNA(Gly) T10003C mutation in mitochondrial haplogroup M11b in a Chinese family with diabetes decreases the steady-state level of tRNA(Gly), increases aberrant reactive oxygen species production, and reduces mitochondrial membrane potential.

Authors:  Wei Li; Chaowei Wen; Weixing Li; Hailing Wang; Xiaomin Guan; Wanlin Zhang; Wei Ye; Jianxin Lu
Journal:  Mol Cell Biochem       Date:  2015-07-02       Impact factor: 3.396

Review 4.  Acrocyanosis: the Flying Dutchman.

Authors:  Andrew K Kurklinsky; Virginia M Miller; Thom W Rooke
Journal:  Vasc Med       Date:  2011-03-22       Impact factor: 3.239

Review 5.  Gastrointestinal and hepatic manifestations of mitochondrial disorders.

Authors:  Shamima Rahman
Journal:  J Inherit Metab Dis       Date:  2013-05-15       Impact factor: 4.982

6.  Functional consequences of mitochondrial tRNA Trp and tRNA Arg mutations causing combined OXPHOS defects.

Authors:  Paulien Smits; Sandy Mattijssen; Eva Morava; Mariël van den Brand; Frans van den Brandt; Frits Wijburg; Ger Pruijn; Jan Smeitink; Leo Nijtmans; Richard Rodenburg; Lambert van den Heuvel
Journal:  Eur J Hum Genet       Date:  2009-10-07       Impact factor: 4.246

7.  Heteroplasmic mutation in the anticodon-stem of mitochondrial tRNA(Val) causing MNGIE-like gastrointestinal dysmotility and cachexia.

Authors:  Rita Horváth; Andreas Bender; Angela Abicht; Elke Holinski-Feder; Birgit Czermin; Tobias Trips; Peter Schneiderat; Hanns Lochmüller; Thomas Klopstock
Journal:  J Neurol       Date:  2009-03-01       Impact factor: 4.849

8.  Molecular pathomechanisms and cell-type-specific disease phenotypes of MELAS caused by mutant mitochondrial tRNA(Trp).

Authors:  Hideyuki Hatakeyama; Ayako Katayama; Hirofumi Komaki; Ichizo Nishino; Yu-Ichi Goto
Journal:  Acta Neuropathol Commun       Date:  2015-08-22       Impact factor: 7.801

9.  Case Report: Mitochondrial Encephalomyopathy Presents as Epilepsy, Ataxia, and Dystonia With a Rare Mutation in MT-TW.

Authors:  Shuang Wang; Jing Miao; Jiachun Feng
Journal:  Front Neurol       Date:  2021-07-01       Impact factor: 4.003

10.  Early Onset and Severe Clinical Course Associated with the m.5540G>A Mutation in MT-TW.

Authors:  Jorge L Granadillo; Timothy Moss; Richard A Lewis; Elise G Austin; Howard Kelfer; Jing Wang; Lee-Jun C Wong; Fernando Scaglia
Journal:  Mol Genet Metab Rep       Date:  2014
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