Literature DB >> 21704194

New mitochondrial tRNA HIS mutation in a family with lactic acidosis and stroke-like episodes (MELAS).

Maria Antonietta Calvaruso1, Michel A Willemsen, Richard J Rodenburg, Mariël van den Brand, Jan A M Smeitink, Leo Nijtmans.   

Abstract

We report a new mutation in m.12146 A>G in the mt-tRNA(His) in a family with a remarkable clinical history having different degrees of lactic acidosis and stroke-like episodes. Biochemical measurements of a muscle biopsy established an isolated complex IV deficiency, while similar analysis of fibroblasts showed a combined complex I,III and IV deficiency. Transmitochondrial cybrid analysis proved that this tRNA(His) mutation causes the enzymatic deficiency. This family illustrates the complexity of the clinical, biochemical and genetic characteristics of a novel mtDNA encoded disorder, as well as the challenge to prove its pathogenicity.
Copyright © 2011 © Elsevier B.V. and Mitochondria Research Society. All rights reserved. Published by Elsevier B.V. All rights reserved.

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Year:  2011        PMID: 21704194     DOI: 10.1016/j.mito.2011.06.004

Source DB:  PubMed          Journal:  Mitochondrion        ISSN: 1567-7249            Impact factor:   4.160


  6 in total

Review 1.  Emerging roles of tRNA in adaptive translation, signalling dynamics and disease.

Authors:  Sebastian Kirchner; Zoya Ignatova
Journal:  Nat Rev Genet       Date:  2014-12-23       Impact factor: 53.242

2.  Mitochondrial Nuclear Retrograde Regulator 1 (MNRR1) rescues the cellular phenotype of MELAS by inducing homeostatic mechanisms.

Authors:  Siddhesh Aras; Neeraja Purandare; Stephanie Gladyck; Mallika Somayajulu-Nitu; Kezhong Zhang; Douglas C Wallace; Lawrence I Grossman
Journal:  Proc Natl Acad Sci U S A       Date:  2020-11-30       Impact factor: 11.205

Review 3.  Molecular and neurological features of MELAS syndrome in paediatric patients: A case series and review of the literature.

Authors:  Lydia M Seed; Andrew Dean; Deepa Krishnakumar; Poe Phyu; Rita Horvath; Pooja Devi Harijan
Journal:  Mol Genet Genomic Med       Date:  2022-04-26       Impact factor: 2.473

4.  l-Arginine in Mitochondrial Encephalopathy, Lactic Acidosis, and Stroke-like Episodes: A Systematic Review.

Authors:  Renae J Stefanetti; Yi Shiau Ng; Linda Errington; Alasdair P Blain; Robert McFarland; Gráinne S Gorman
Journal:  Neurology       Date:  2022-04-15       Impact factor: 11.800

Review 5.  The molecular pathology of pathogenic mitochondrial tRNA variants.

Authors:  Uwe Richter; Robert McFarland; Robert W Taylor; Sarah J Pickett
Journal:  FEBS Lett       Date:  2021-02-12       Impact factor: 3.864

Review 6.  Transfer RNA and human disease.

Authors:  Jamie A Abbott; Christopher S Francklyn; Susan M Robey-Bond
Journal:  Front Genet       Date:  2014-06-03       Impact factor: 4.599

  6 in total

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