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Neonatal mitochondrial hepatoencephalopathy caused by novel GFM1 mutations.

Authors :
Ravn K
Schönewolf-Greulich B
Hansen RM
Bohr AH
Duno M
Wibrand F
Ostergaard E
Source :
Molecular genetics and metabolism reports [Mol Genet Metab Rep] 2015 Feb 20; Vol. 3, pp. 5-10. Date of Electronic Publication: 2015 Feb 20 (Print Publication: 2015).
Publication Year :
2015

Abstract

Disorders caused by defects in the mitochondrial translation system are clinically and genetically heterogeneous. The elongation phase of mitochondrial protein synthesis requires, among many other components, three nuclear-encoded elongation factors: EFTu (TUFM; 602389), EFTs (TSFM; 604723), and EFG1 (GFM1; 606639). Mutations have been identified in the genes encoding all three elongation factors, and they result in combined respiratory chain deficiencies and severe phenotypes with an early fatal outcome. So far, only eleven patients have been reported with mutations in GFM1. Here we describe an additional three patients with novel GFM1 mutations. Our results confirm the tissue-specific effect of GFM1 mutations, since we found only slightly decreased respiratory chain enzyme activities in muscle and fibroblasts, but a severe deficiency in the liver. Hence, a thorough biochemical evaluation is important to guide genetic investigation in patients suspected for a mitochondrial disorder.

Details

Language :
English
ISSN :
2214-4269
Volume :
3
Database :
MEDLINE
Journal :
Molecular genetics and metabolism reports
Publication Type :
Academic Journal
Accession number :
26937387
Full Text :
https://doi.org/10.1016/j.ymgmr.2015.01.004