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
References (28)
28 references, click to expand
-
Replication of animal mitochondrial DNA.
Cell. 1982 Apr;28(4):693-705
PMID: 6178513
-
Sequence and gene organization of mouse mitochondrial DNA.
Cell. 1981 Oct;26(2 Pt 2):167-80
PMID: 7332926
-
A technique for radiolabeling DNA restriction endonuclease fragments to high specific activity.
Anal Biochem. 1983 Jul 1;132(1):6-13
PMID: 6312838
-
Mitochondrial DNA polymorphism in Japanese. I. Analysis with restriction enzymes of six base pair recognition.
Hum Genet. 1984;68(4):324-32
PMID: 6096257
-
Six unidentified reading frames of human mitochondrial DNA encode components of the respiratory-chain NADH dehydrogenase.
Nature. 1985 Apr 18-24;314(6012):592-7
PMID: 3921850
-
Maternally inherited mitochondrial myopathy and myoclonic epilepsy.
Ann Neurol. 1985 Mar;17(3):228-37
PMID: 3922281
-
Mitochondrial myopathies.
Ann Neurol. 1985 Jun;17(6):521-38
PMID: 3927817
-
The retinal manifestations of mitochondrial myopathy. A study of 22 cases.
Arch Ophthalmol. 1985 Dec;103(12):1825-30
PMID: 4074172
-
Mitochondrial DNA polymorphism in Japanese. II. Analysis with restriction enzymes of four or five base pair recognition.
Hum Genet. 1986 Feb;72(2):105-17
PMID: 3002958
-
Human mitochondrial DNA types in two Israeli populations--a comparative study at the DNA level.
Am J Hum Genet. 1986 Mar;38(3):341-51
PMID: 3006483
-
Genetic studies on the Tharu population of Nepal: restriction endonuclease polymorphisms of mitochondrial DNA.
Am J Hum Genet. 1986 Oct;39(4):502-12
PMID: 2876631
-
The clinical features of mitochondrial myopathy.
Brain. 1986 Oct;109 ( Pt 5):915-38
PMID: 3779373
-
Mitochondrial DNA and human evolution.
Nature. 1987 Jan 1-7;325(6099):31-6
PMID: 3025745
-
Mitochondrial encephalomyopathy. Association with an NADH dehydrogenase deficiency.
Arch Neurol. 1987 Jul;44(7):775-8
PMID: 3593065
-
Primer-directed enzymatic amplification of DNA with a thermostable DNA polymerase.
Science. 1988 Jan 29;239(4839):487-91
PMID: 2448875
-
Deletions of muscle mitochondrial DNA in patients with mitochondrial myopathies.
Nature. 1988 Feb 25;331(6158):717-9
PMID: 2830540
-
Mitochondrial DNA polymorphisms in Italy. I. Population data from Sardinia and Rome.
Ann Hum Genet. 1986 Oct;50(Pt 4):327-38
PMID: 2831789
-
Deletions of mitochondrial DNA in Kearns-Sayre syndrome.
Neurology. 1988 Sep;38(9):1339-46
PMID: 3412580
-
Nucleotide sequence and gene organization of sea urchin mitochondrial DNA.
J Mol Biol. 1988 Jul 20;202(2):185-217
PMID: 3172215
-
Mitochondrial DNA mutation associated with Leber's hereditary optic neuropathy.
Science. 1988 Dec 9;242(4884):1427-30
PMID: 3201231
-
Duplications of mitochondrial DNA in mitochondrial myopathy.
Lancet. 1989 Feb 4;1(8632):236-40
PMID: 2563411
-
Mitochondrial DNA deletions in progressive external ophthalmoplegia and Kearns-Sayre syndrome.
N Engl J Med. 1989 May 18;320(20):1293-9
PMID: 2541333
-
Genetic heterogeneity and mitochondrial DNA heteroplasmy in Leber's hereditary optic neuropathy.
J Med Genet. 1989 Dec;26(12):739-43
PMID: 2575667
-
DNA sequencing with chain-terminating inhibitors.
Proc Natl Acad Sci U S A. 1977 Dec;74(12):5463-7
PMID: 271968
-
Conservation of genes coding for proteins synthesized in human mitochondria.
Somatic Cell Genet. 1978 Nov;4(6):633-45
PMID: 741350
-
Maternal inheritance of human mitochondrial DNA.
Proc Natl Acad Sci U S A. 1980 Nov;77(11):6715-9
PMID: 6256757
-
Sequence and organization of the human mitochondrial genome.
Nature. 1981 Apr 9;290(5806):457-65
PMID: 7219534
-
Mitochondrial DNA polymorphism in a maternal lineage of Holstein cows.
Proc Natl Acad Sci U S A. 1982 Aug;79(15):4686-90
PMID: 6289312