Document Detail


Metabolism of the cholestanol precursor cholesta-4,6-dien-3-one in different tissues.
MedLine Citation:
PMID:  3676336     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Cerebrotendinous xanthomatosis (CTX) is a lipid storage disease where the basic defect is a lack of the mitochondrial C27-steroid 26-hydroxylase involved in bile acid synthesis (EC 1.14.13.15). Cholestanol and cholesterol accumulate in all tissues. At least part of the accumulation of cholestanol is due to a 7 alpha-dehydroxylation of early bile acid intermediates. Cholesta-4,6-dien-3-one, a proposed intermediate in this pathway, is found in increased concentrations in serum of the patients. This study shows that cholesta-4,6-dien-3-one may be metabolized to 4-cholesten-3-one and cholestanol by liver, adrenals and brain. No conversion was found in intestinal mucosa or in kidneys. The capacity to convert cholesta-4,6-dien-3-one into 4-cholesten-3-one and cholestanol varied in different tissues as well as in different species. The results are discussed in relation to the cholestanol accumulation in CTX.
Authors:
M S Buchmann; I Björkhem; S Skrede
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Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't    
Journal Detail:
Title:  Biochimica et biophysica acta     Volume:  922     ISSN:  0006-3002     ISO Abbreviation:  Biochim. Biophys. Acta     Publication Date:  1987 Nov 
Date Detail:
Created Date:  1987-12-28     Completed Date:  1987-12-28     Revised Date:  2006-11-15    
Medline Journal Info:
Nlm Unique ID:  0217513     Medline TA:  Biochim Biophys Acta     Country:  NETHERLANDS    
Other Details:
Languages:  eng     Pagination:  111-7     Citation Subset:  IM    
Affiliation:
Institute of Clinical Biochemistry, Rikshospitalet, University of Oslo, Norway.
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MeSH Terms
Descriptor/Qualifier:
Animals
Cholestanols / biosynthesis*
Cholestenes / metabolism*
Cholestenones / metabolism*
Chromatography, High Pressure Liquid
Female
Humans
Kinetics
Microsomes / metabolism*
Microsomes, Liver / metabolism
Organ Specificity
Rats
Rats, Inbred Strains
Tritium
Chemical
Reg. No./Substance:
0/Cholestanols; 0/Cholestenes; 0/Cholestenones; 10028-17-8/Tritium; 566-93-8/4,6-cholestadien-3-one

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


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