Literature DB >> 9262546

Macular pattern retinal dystrophy, adult-onset diabetes, and deafness: a family study of A3243G mitochondrial heteroplasmy.

T J Harrison1, R G Boles, D R Johnson, C LeBlond, L J Wong.   

Abstract

PURPOSE: To correlate mitochondrial DNA (mtDNA) mutation with phenotypic expression in three members of a Finnish family with macroreticular pattern dystrophy, non-insulin-dependent diabetes mellitus, and deafness.
METHODS: A multiplex polymerase chain reaction/allele-specific oligonucleotide method was used to screen 10 mtDNA point mutations known to cause mitochondrial DNA disorders, often characterized by myopathy, retinopathy, or both. Quantitative analysis of mutant mitochondrial DNA was performed in three tissue types in each of three family members by determining the percentage of mutant mtDNA in blood, buccal cells, and hair follicles.
RESULTS: A heteroplasmic A3243G mtDNA point mutation was found in each of the three family members studied. Heteroplasmy refers to the coexistence of normal and mutant mitochondria in the same cell. The average percentage of mutant heteroplasmy ranged from 11% to 25%. The severity of disease symptoms did not appear to correlate with the average degree of mutant heteroplasmy in the three tissues analyzed.
CONCLUSIONS: Molecular confirmation in this family emphasizes the importance of mitochondrial DNA mutation analysis in patients with macular pattern retinal dystrophy and other mitochondrial associated nonocular disease, such as non-insulin-dependent diabetes mellitus and deafness. The detection of a disease-associated mitochondrial DNA mutation warrants genetic counseling, appropriate patient follow-up, and possibly the molecular testing of other at-risk family members.

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Year:  1997        PMID: 9262546     DOI: 10.1016/s0002-9394(14)70787-1

Source DB:  PubMed          Journal:  Am J Ophthalmol        ISSN: 0002-9394            Impact factor:   5.258


  8 in total

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Review 2.  Genotype-phenotype correlations and differential diagnosis in autosomal dominant macular disease.

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4.  Audiologic and genetic features of the A3243G mtDNA mutation.

Authors:  Richard J Vivero; Xiaomei Ouyang; Yeunjung Grant Kim; Wendy Liu; Lilin Du; Denise Yan; Xue Zhong Liu
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5.  [Retinal pigment epithelium atrophy and hypacousia in monozygotic twin sisters].

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6.  Genetic linkage analysis of a novel syndrome comprising North Carolina-like macular dystrophy and progressive sensorineural hearing loss.

Authors:  P J Francis; S Johnson; B Edmunds; R E Kelsell; E Sheridan; C Garrett; G E Holder; D M Hunt; A T Moore
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7.  Rapid Detection of the mt3243A > G Mutation Using Urine Sediment in Elderly Chinese Type 2 Diabetic Patients.

Authors:  Yinan Zhang; Xiujuan Du; Xinqian Geng; Chen Chu; Huijuan Lu; Yixie Shen; Ruihua Chen; Pingyan Fang; Yanmei Feng; Xiaojie Zhang; Yan Chen; Yanping Zhou; Congrong Wang; Weiping Jia
Journal:  J Diabetes Res       Date:  2017-06-21       Impact factor: 4.011

8.  Characterisation of the macular dystrophy in patients with the A3243G mitochondrial DNA point mutation with fundus autofluorescence.

Authors:  P P Rath; S Jenkins; M Michaelides; A Smith; M G Sweeney; M B Davis; F W Fitzke; A C Bird
Journal:  Br J Ophthalmol       Date:  2008-05       Impact factor: 4.638

  8 in total

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