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Unusual adult-onset Leigh syndrome presentation due to the mitochondrial m.9176T>C mutation

Authors :
Marco Mattioli
Elisa Fassone
M. Servida
Alessandra Cosi
Mafalda Rizzuti
Andreina Bordoni
Alessio Di Fonzo
Martina Collotta
Nereo Bresolin
Stefania Corti
Monica Sciacco
Valeria Lucchini
Dario Ronchi
Giacomo P. Comi
Maurizio Moggio
Marco Ronzoni
Source :
Biochemical and Biophysical Research Communications. 412:245-248
Publication Year :
2011
Publisher :
Elsevier BV, 2011.

Abstract

Leigh syndrome (LS) is an incurable, nearly always fatal, neurodegenerative, pediatric disorder that results from respiratory chain failure. The most common mitochondrial DNA (mtDNA) mutations that result in LS are m.8993T→C/G and m.9176T→C/G, which were previously found in several patients with early-onset Leigh syndrome. Here, we describe clinical and molecular features of a novel pedigree, where LS developed in two siblings. The proband was a young woman with an unusual adult-onset LS. She harbored a homoplasmic m.9176T→C mutation, based on analysis of a muscle biopsy. In contrast, the brother died at a young age. This novel case report and literature review highlights the variability of phenotypic expression of the m.9176T→C mutation.

Details

ISSN :
0006291X
Volume :
412
Database :
OpenAIRE
Journal :
Biochemical and Biophysical Research Communications
Accession number :
edsair.doi.dedup.....4bdb977a35e09478c634ad1410fc6ee1
Full Text :
https://doi.org/10.1016/j.bbrc.2011.07.076