| Literature DB >> 34590675 |
Nathaniel K Mullin1, Kristin R Anfinson1, Megan J Riker1, Kelsey L Wieland1, Nicole J Tatro1, Todd E Scheetz1, Robert F Mullins1, Edwin M Stone1, Budd A Tucker1.
Abstract
The m.3243A>G mutation in the mitochondrial genome commonly causes retinal degeneration in patients with maternally inherited diabetes and deafness and mitochondrial encephalopathy, lactic acidosis and stroke-like episodes. Like other mitochondrial mutations, m.3243A>G is inherited from the mother with a variable proportion of wild type and mutant mitochondrial genomes in different cells. The mechanism by which the m.3243A>G variant in each tissue relates to the manifestation of disease phenotype is not fully understood. Using a digital PCR assay, we found that the % m.3243G in skin derived dermal fibroblasts was positively correlated with that of blood from the same individual. The % m.3243G detected in fibroblast cultures remained constant over multiple passages and was negatively correlated with mtDNA copy number. Although the % m.3243G present in blood was not correlated with severity of vision loss, as quantified by Goldmann visual field, a significant negative correlation between % m.3243G and the age of onset of visual symptoms was detected. Altogether, these results indicate that precise measurement of % m.3243G in clinically accessible tissues such as skin and blood may yield information relevant to the management of retinal m.3243A>G-associated disease.Entities:
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Year: 2022 PMID: 34590675 PMCID: PMC8895728 DOI: 10.1093/hmg/ddab289
Source DB: PubMed Journal: Hum Mol Genet ISSN: 0964-6906 Impact factor: 5.121