Literature DB >> 34590675

Sensitive quantification of m.3243A>G mutational proportion in non-retinal tissues and its relationship with visual symptoms.

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.
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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


  37 in total

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Authors:  Teun M Klein Gunnewiek; Eline J H Van Hugte; Monica Frega; Gemma Solé Guardia; Katharina Foreman; Daan Panneman; Britt Mossink; Katrin Linda; Jason M Keller; Dirk Schubert; David Cassiman; Richard Rodenburg; Noemi Vidal Folch; Devin Oglesbee; Ester Perales-Clemente; Timothy J Nelson; Eva Morava; Nael Nadif Kasri; Tamas Kozicz
Journal:  Cell Rep       Date:  2020-04-21       Impact factor: 9.423

Review 2.  Mitochondrial DNA genetics and the heteroplasmy conundrum in evolution and disease.

Authors:  Douglas C Wallace; Dimitra Chalkia
Journal:  Cold Spring Harb Perspect Biol       Date:  2013-11-01       Impact factor: 10.005

3.  Relationship of genotype to phenotype in fibroblast-derived transmitochondrial cell lines carrying the 3243 mutation associated with the MELAS encephalomyopathy: shift towards mutant genotype and role of mtDNA copy number.

Authors:  H A Bentlage; G Attardi
Journal:  Hum Mol Genet       Date:  1996-02       Impact factor: 6.150

4.  Clinical Phenotype and Segregation of Mitochondrial 3243A>G Mutation in 2 Pairs of Monozygotic Twins.

Authors:  Kengo Maeda; Hiromichi Kawai; Mitsuru Sanada; Tomoya Terashima; Nobuhiro Ogawa; Ryo Idehara; Tetsuya Makiishi; Hitoshi Yasuda; Shun-Ichi Sato; Ken-Ichi Hoshi; Hiroyuki Yahikozawa; Katsuji Nishi; Yasushi Itoh; Kazumasa Ogasawara; Kazuo Tomita; Hiroko P Indo; Hideyuki J Majima
Journal:  JAMA Neurol       Date:  2016-08-01       Impact factor: 18.302

5.  Organization of multiple nucleoids and DNA molecules in mitochondria of a human cell.

Authors:  M Satoh; T Kuroiwa
Journal:  Exp Cell Res       Date:  1991-09       Impact factor: 3.905

6.  Human iPSC disease modelling reveals functional and structural defects in retinal pigment epithelial cells harbouring the m.3243A > G mitochondrial DNA mutation.

Authors:  Valeria Chichagova; Dean Hallam; Joseph Collin; Adriana Buskin; Gabriele Saretzki; Lyle Armstrong; Patrick Yu-Wai-Man; Majlinda Lako; David H Steel
Journal:  Sci Rep       Date:  2017-09-26       Impact factor: 4.379

7.  Differential macular and peripheral expression of bestrophin in human eyes and its implication for best disease.

Authors:  Robert F Mullins; Markus H Kuehn; Elizabeth A Faidley; Nasreen A Syed; Edwin M Stone
Journal:  Invest Ophthalmol Vis Sci       Date:  2007-07       Impact factor: 4.799

8.  Computerized quantitative analysis of kinetic visual fields.

Authors:  R G Weleber; W R Tobler
Journal:  Am J Ophthalmol       Date:  1986-04-15       Impact factor: 5.258

9.  Volume of visual field assessed with kinetic perimetry and its application to static perimetry.

Authors:  John B Christoforidis
Journal:  Clin Ophthalmol       Date:  2011-04-26

10.  Mitochondrial DNA heteroplasmy in human health and disease.

Authors:  George B Stefano; Richard M Kream
Journal:  Biomed Rep       Date:  2016-02-04
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  1 in total

1.  In Reply to "The Spectrum of Renal Abnormalities in Mitochondrial Disorders Is Broad".

Authors:  Toshiyuki Imasawa; Kei Murayama
Journal:  Kidney Int Rep       Date:  2022-05-21
  1 in total

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