Document Detail


Two Japanese brothers with hereditary gamma-glutamyl transpeptidase deficiency.
MedLine Citation:
PMID:  15702405     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
We report on two Japanese brothers with hereditary deficiency in gamma-glutamyl transpeptidase. The propositus was 48 years old when he first visited our medical center and had a 51-year-old brother. The brothers were both tall and slender and had long limbs; the younger was diagnosed as having Marfan syndrome. Both patients both showed a tendency to retarded mental development. gamma-Glutamyltranspeptidase activity was below the detection limit of 1 IU/L in both patients. Glutathionaemia and glutathionuria were evident in both brothers. The analyses of sulphydryl compounds in the plasma (and serum for certain test items) and urine indicated high concentrations of glutathione, gamma-glutamylcysteine, cysteine and cysteinylglycine. Urine amino acid analysis on an automatic analyser showed a slightly increased excretion of cystine and a large peak in the citrulline position due, at least in part, to thio-compounds.
Authors:
M Iida; T Yasuhara; H Mochizuki; H Takakura; T Yanagisawa; H Kubo
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Journal of inherited metabolic disease     Volume:  28     ISSN:  0141-8955     ISO Abbreviation:  J. Inherit. Metab. Dis.     Publication Date:  2005  
Date Detail:
Created Date:  2005-02-09     Completed Date:  2005-06-07     Revised Date:  2007-03-21    
Medline Journal Info:
Nlm Unique ID:  7910918     Medline TA:  J Inherit Metab Dis     Country:  Netherlands    
Other Details:
Languages:  eng     Pagination:  49-55     Citation Subset:  IM    
Affiliation:
Laboratory, Saitama Cardiovascular and Respiratory Center, Saitama, Japan. a0135785@pref.saitama.lg.jp
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MeSH Terms
Descriptor/Qualifier:
Amino Acid Metabolism, Inborn Errors / diagnosis,  genetics
Amino Acids / urine
Borohydrides / chemistry
Chromatography, High Pressure Liquid
Cysteine / metabolism
Dipeptides / chemistry
Family Health
Glutathione / metabolism,  urine
Humans
Japan
Male
Marfan Syndrome / complications
Middle Aged
Siblings
Time Factors
gamma-Glutamyltransferase / biosynthesis,  deficiency*
Chemical
Reg. No./Substance:
0/Amino Acids; 0/Borohydrides; 0/Dipeptides; 52-90-4/Cysteine; 70-18-8/Glutathione; 70555-24-7/cystinylglycine; EC 2.3.2.2/gamma-Glutamyltransferase

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


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