| Literature DB >> 21704194 |
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.Entities:
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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