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PMID: 7798979 Published · ppublish English Case Reports Clinical Trial Journal Article Research Support, Non-U.S. Gov't

Brain and muscle energy metabolism studied in vivo by 31P-magnetic resonance spectroscopy in NARP syndrome.

Journal of neurology, neurosurgery, and psychiatry ·Vol. 57 ·No. 12 ·1994-12-00 ·Pages 1492-6

Lodi R, Montagna P, Iotti S, Zaniol P, Barboni P, Puddu P, Barbiroli B

Abstract

Phosphorus magnetic resonance spectroscopy (31P-MRS) was used to study in vivo the energy metabolism of brain and skeletal muscle in two members of an Italian pedigree with NARP syndrome due to a point mutation at bp 8993 of mtDNA. In the youngest patient, a 13 year old girl with retinitis pigmentosa, ataxia, and psychomotor retardation, there was an alteration of brain energy metabolism shown by a decreased phosphocreatine content, increased [ADP] and decreased phosphorylation potential. The energy metabolism of her skeletal muscle was also abnormal, as shown by resting higher inorganic phosphate and lower phosphocreatine concentrations than in normal subjects. Her mother, a 41 year old woman with minimal clinical involvement, showed a milder derangement of brain energy metabolism and normal skeletal muscle. Findings with MRS showed that this point mutation of mtDNA is responsible for a derangement of energy metabolism in skeletal muscle and even more so in the brain.

MeSH Terms
Adenosine Diphosphate/analysis Adolescent Adult Ataxia/complications,genetics,metabolism Brain/metabolism Brain Chemistry Case-Control Studies DNA, Mitochondrial/genetics Energy Metabolism Female Follow-Up Studies Humans Intellectual Disability/complications,genetics,metabolism Magnetic Resonance Spectroscopy Muscles/chemistry,metabolism Pedigree Phosphocreatine/analysis Phosphorus Isotopes Phosphorylation Point Mutation/genetics Retinitis Pigmentosa/complications,genetics,metabolism Syndrome
Chemicals
DNA, Mitochondrial Phosphorus Isotopes Phosphocreatine Adenosine Diphosphate
Authors & Affiliations
7 authors, click to expand affiliations / ORCID
Lodi R
Cattedra di Biochimica Clinica, Istituto di Patologia Medica D Campanacci, Bologna, Italy.
Montagna P
Iotti S
Zaniol P
Barboni P
Puddu P
Barbiroli B
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Article Info
Journal
Journal of neurology, neurosurgery, and psychiatry
Abbr.
J Neurol Neurosurg Psychiatry
ISSN
0022-3050
Published
1994-12-00
Pages
1492-6
Language
English
Region
England
NLM ID
2985191R
PMCID
PMC1073231
Subset
IM
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