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PMID: 2137962 Published · ppublish English Case Reports Journal Article Research Support, Non-U.S. Gov't

A new mitochondrial disease associated with mitochondrial DNA heteroplasmy.

American journal of human genetics ·Vol. 46 ·No. 3 ·1990-03-00 ·Pages 428-33

Holt IJ, Harding AE, Petty RK, Morgan-Hughes JA

Abstract

A variable combination of developmental delay, retinitis pigmentosa, dementia, seizures, ataxia, proximal neurogenic muscle weakness, and sensory neuropathy occurred in four members of a family and was maternally transmitted. There was no histochemical evidence of mitochondrial myopathy. Blood and muscle from the patients contained two populations of mitochondrial DNA, one of which had a previously unreported restriction site for AvaI. Sequence analysis showed that this was due to a point mutation at nucleotide 8993, resulting in an amino acid change from a highly conserved leucine to arginine in subunit 6 of mitochondrial H(+)-ATPase. There was some correlation between clinical severity and the amount of mutant mitochondrial DNA in the patients; this was present in only small quantities in the blood of healthy elderly relatives in the same maternal line.

MeSH Terms
Adult Aged Aged, 80 and over Amino Acids/genetics Animals Base Sequence Child, Preschool DNA, Mitochondrial/genetics Deoxyribonucleases, Type II Site-Specific Female Humans Male Middle Aged Mitochondria, Muscle/enzymology,metabolism Molecular Sequence Data Muscular Diseases/enzymology,genetics,pathology Mutation Pedigree Proton-Translocating ATPases/blood,genetics,metabolism
Chemicals
Amino Acids DNA, Mitochondrial CYCGRG-specific type II deoxyribonucleases Deoxyribonucleases, Type II Site-Specific Proton-Translocating ATPases
Authors & Affiliations
4 authors, click to expand affiliations / ORCID
Holt I J
University Department of Clinical Neurology, Institute of Neurology, London, England.
Harding A E
Petty R K
Morgan-Hughes J A
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Article Info
Journal
American journal of human genetics
Abbr.
Am J Hum Genet
ISSN
0002-9297
Published
1990-03-00
Pages
428-33
Language
English
Region
United States
NLM ID
0370475
PMCID
PMC1683641
Subset
IM
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