Document Detail


Fatal infantile mitochondrial encephalomyopathy with complex I and IV deficiencies.
MedLine Citation:
PMID:  8388688     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
A 4-month-old male infant had a fatal infantile mitochondrial disease associated with cardiomyopathy. He had elevated lactate concentrations in blood and cerebrospinal fluid and an increased lactate/pyruvate ratio. Histochemical analysis of muscle biopsy revealed several ragged-red fibers on modified Gomori trichrome stain and mildly decreased cytochrome c oxidase (complex IV) activity. Complex I and IV activities of the respiratory chain in muscle were decreased to about 35% of normal values biochemically; subunits of the two complexes were decreased nonselectively on immunoblotting. Mitochondrial DNA analysis failed to detect any mutation. Complex I and IV deficiencies should be considered as one of the causes of fatal infantile mitochondrial disease.
Authors:
T Nagai; Y Tuchiya; Y Taguchi; R Sakuta; T Ichiki; I Nonaka
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Pediatric neurology     Volume:  9     ISSN:  0887-8994     ISO Abbreviation:  Pediatr. Neurol.     Publication Date:    1993 Mar-Apr
Date Detail:
Created Date:  1993-07-01     Completed Date:  1993-07-01     Revised Date:  2007-11-15    
Medline Journal Info:
Nlm Unique ID:  8508183     Medline TA:  Pediatr Neurol     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  151-4     Citation Subset:  IM    
Affiliation:
Division of Ultrastructural Research; National Institute of Neuroscience, Tokyo, Japan.
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MeSH Terms
Descriptor/Qualifier:
Acidosis, Lactic / genetics,  pathology*
Amino Acids / blood
Biopsy
Carnitine / deficiency
Cytochrome-c Oxidase Deficiency*
Follow-Up Studies
Humans
Infant
Infant, Newborn
Male
Microscopy, Electron
Mitochondrial Encephalomyopathies / genetics,  pathology*
Muscles / pathology
NAD(P)H Dehydrogenase (Quinone) / deficiency*
Neurologic Examination
Chemical
Reg. No./Substance:
0/Amino Acids; 541-15-1/Carnitine; EC 1.6.5.2/NAD(P)H Dehydrogenase (Quinone)

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