Literature DB >> 1141769

Identification of pentahydroxy bile alcohols in cerebrotendinous xanthomatosis: characterization of 5beta-cholestane-3alpha, 7alpha, 12alpha, 24xi, 25-pentol and 5beta-cholestane-3alpha, 7alpha, 12alpha, 23xi, 25-pentol.

S Shefer, B Dayal, G S Tint, G Salen, E H Mosbach.   

Abstract

This paper describes studies dealing with the nature of the C27 pentahydroxy bile alcohols present in the bile and feces of two patients with cerebrotendinous xanthomatosis (CTX). The presence of a bile alcohol having the structure 5beta-cholestane-3alpha,7alpha,12alpha,24alpha,25-pentol was confirmed by separation of the two 24-hydroxy epimers of 5beta-cholestane-3alpha,7alpha,12alpha,24,25-pentol and characterization of the dpimers by gas-liquid chromatography and infrared and mass spectrometry. Tentative assignment of the 24alpha and 24beta configuration was made on the basis of molecular rotation differences. A second major bile alcohol excreted by the CTX subjects was 5beta-cholestane-3alpha,7alpha,12alpha,23xi,25-pentol. Its structure was determined by infrared spectrometry, proton magnetic resonance spectrometry, and mass spectrometry because a reference compound was not available.

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Year:  1975        PMID: 1141769

Source DB:  PubMed          Journal:  J Lipid Res        ISSN: 0022-2275            Impact factor:   5.922


  6 in total

Review 1.  Cerebrotendinous xanthomatosis: clinical, electrophysiological and nerve biopsy findings, and response to treatment with chenodeoxycholic acid.

Authors:  M Donaghy; R H King; R O McKeran; M S Schwartz; P K Thomas
Journal:  J Neurol       Date:  1990-06       Impact factor: 4.849

2.  Cholic acid biosynthesis: the enzymatic defect in cerebrotendinous xanthomatosis.

Authors:  G Salen; S Shefer; F W Cheng; B Dayal; A K Batta; G S Tint
Journal:  J Clin Invest       Date:  1979-01       Impact factor: 14.808

3.  Bile acid synthesis in man. In vivo activity of the 25-hydroxylation pathway.

Authors:  W C Duane; P A Pooler; J N Hamilton
Journal:  J Clin Invest       Date:  1988-07       Impact factor: 14.808

4.  A 25-hydroxylation pathway of cholic acid biosynthesis in man and rat.

Authors:  S Shefer; F W Cheng; B Dayal; S Hauser; G S Tint; G Salen; E H Mosbach
Journal:  J Clin Invest       Date:  1976-04       Impact factor: 14.808

5.  Biosynthesis of bile acids in cerebrotendinous xanthomatosis. Relationship of bile acid pool sizes and synthesis rates to hydroxylations at C-12, C-25, and C-26.

Authors:  G Salen; S Shefer; G S Tint; G Nicolau; B Dayal; A K Batta
Journal:  J Clin Invest       Date:  1985-08       Impact factor: 14.808

6.  Cerebrotendinous xanthomatosis: a review of biochemical findings of the patient population in The Netherlands.

Authors:  B J Koopman; B G Wolthers; J C van der Molen; W van der Slik; R J Waterreus; A van Spreeken
Journal:  J Inherit Metab Dis       Date:  1988       Impact factor: 4.982

  6 in total

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