Literature DB >> 23206802

Different laboratory and muscle biopsy findings in a family with an m.8851T>C mutation in the mitochondrial MTATP6 gene.

Tomas Honzik1, Marketa Tesarova, Kamila Vinsova, Hana Hansikova, Martin Magner, Hana Kratochvilova, Josef Zamecnik, Jiri Zeman, Pavel Jesina.   

Abstract

We report the second known family with a very rare, maternally inherited missense m.8851T>C mutation in the mitochondrial MTATP6 gene. A failure to thrive, microcephaly, psychomotor retardation and hypotonia were present in a 3-year-old girl with a high mtDNA mutation load (87-97%). Ataxia and Leigh syndrome were subsequently documented in a neurological examination and brain MRI. A muscle biopsy demonstrated decreased ATP synthase and an accumulation of succinate dehydrogenase products, indicating mitochondrial myopathy. Her 36-year-old mother (68% blood heteroplasmy) developed peripheral neuropathy and muscle weakness at the age of 22 years. Our findings extend the clinical and laboratory phenotype associated with the m.8851T>C mutation.
Copyright © 2012 Elsevier Inc. All rights reserved.

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Year:  2012        PMID: 23206802     DOI: 10.1016/j.ymgme.2012.11.002

Source DB:  PubMed          Journal:  Mol Genet Metab        ISSN: 1096-7192            Impact factor:   4.797


  4 in total

1.  Novel two-step derivation method for the synchronous analysis of inherited metabolic disorders using urine.

Authors:  Xiao-Qi Sheng; Yi-Chao Wang
Journal:  Exp Ther Med       Date:  2017-03-02       Impact factor: 2.447

2.  Hyperammonemic crisis in a child with ATP synthase deficiency caused by mtDNA mutation m.8851T>C.

Authors:  Veronika Dvorakova; Martin Magner; Tomas Honzik
Journal:  Mol Genet Metab Rep       Date:  2014-12-18

Review 3.  ATP Synthase Diseases of Mitochondrial Genetic Origin.

Authors:  Alain Dautant; Thomas Meier; Alexander Hahn; Déborah Tribouillard-Tanvier; Jean-Paul di Rago; Roza Kucharczyk
Journal:  Front Physiol       Date:  2018-04-04       Impact factor: 4.566

4.  Mitochondrial DNA Analysis from Exome Sequencing Data Improves Diagnostic Yield in Neurological Diseases.

Authors:  Olivia V Poole; Chiara Pizzamiglio; David Murphy; Micol Falabella; William L Macken; Enrico Bugiardini; Cathy E Woodward; Robyn Labrum; Stephanie Efthymiou; Vincenzo Salpietro; Viorica Chelban; Rauan Kaiyrzhanov; Reza Maroofian; Anthony A Amato; Allison Gregory; Susan J Hayflick; Hallgeir Jonvik; Nicholas Wood; Henry Houlden; Jana Vandrovcova; Michael G Hanna; Alan Pittman; Robert D S Pitceathly
Journal:  Ann Neurol       Date:  2021-04-01       Impact factor: 11.274

  4 in total

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