| Literature DB >> 29266179 |
J H Langdahl1,2,3, M Larsen1,2, M Frost2,4, P H Andersen3, K B Yderstraede5, J Vissing6, M Dunø7, M Thomassen1,2, A L Frederiksen1,2.
Abstract
Carriers of the mitochondrial mutation m.3243A>G presents highly variable phenotypes including mitochondrial encephalomyopathy, lactoacidosis and stroke-like episodes (MELAS). We conducted a follow-up study to evaluate changes in leucocyte heteroplasmy and the clinical phenotypes in m.3243A>G carriers. Leucocyte heteroplasmy was determined by next generation sequencing covered by 100 000X reads in 32 individuals with a median follow-up of 10.2 years. Ten-year clinical follow-up is reported in 46 individuals. The annual leucocyte mutation level declined by -0.7 (±0.4) percentage points/year (P < .0001), and correlated with the level of the initial sample (ρ = -0.92, P < .0001). Eleven of 46 m.3243A>G carriers died and clinical symptoms progressed. This longitudinal study shows the decline in leucocyte m.3243A>G heteroplasmy associates with the level of the initial sample. Further, there was a high mortality among carriers.Entities:
Keywords: MELAS; heteroplasmy; m.3243A>G; mitochondria
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Year: 2018 PMID: 29266179 DOI: 10.1111/cge.13201
Source DB: PubMed Journal: Clin Genet ISSN: 0009-9163 Impact factor: 4.438