Document Detail


3-Methylglutaconic aciduria in the Iraqi-Jewish 'optic atrophy plus' (Costeff) syndrome.
MedLine Citation:
PMID:  7510656     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Eleven new patients of Iraqi-Jewish origin with bilateral optic atrophy, neurological abnormalities ('optic atrophy plus' syndrome) and 3-methylglutaconic aciduria (type III) are described. Clinical abnormalities in decreasing order of frequency were bilateral optic atrophy, extrapyramidal signs, spasticity, ataxia, dysarthria and cognitive deficit. An association with age was found only for spasticity. Spasticity, extrapyramidal signs and optic atrophy frequently led to major disability, in contrast to ataxia, dysarthria and cognitive deficit. The combined excretion of 3-methylglutaconic and 3-methylglutaric acid ranged between 9 and 187 mmol/mol creatinine. The primary enzymatic defect possibly may reside in the mitochondrial respiratory chain.
Authors:
O N Elpeleg; H Costeff; A Joseph; Y Shental; R Weitz; K M Gibson
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Developmental medicine and child neurology     Volume:  36     ISSN:  0012-1622     ISO Abbreviation:  Dev Med Child Neurol     Publication Date:  1994 Feb 
Date Detail:
Created Date:  1994-04-21     Completed Date:  1994-04-21     Revised Date:  2009-11-11    
Medline Journal Info:
Nlm Unique ID:  0006761     Medline TA:  Dev Med Child Neurol     Country:  ENGLAND    
Other Details:
Languages:  eng     Pagination:  167-72     Citation Subset:  IM    
Affiliation:
Metabolic Unit, Shaare Zedek Medical Center, Jerusalem, Israel.
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Adult
Child
Child, Preschool
Cognition Disorders / ethnology,  urine
Female
Glutarates / urine*
Humans
Iraq
Jews*
Male
Meglutol / analogs & derivatives,  urine
Movement Disorders / ethnology*,  urine
Optic Atrophy / ethnology*,  urine
Syndrome
Chemical
Reg. No./Substance:
0/Glutarates; 503-49-1/Meglutol; 5746-90-7/3-methylglutaconic acid; 626-51-7/3-methylglutaric acid

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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