Document Detail


An autopsy case of gallbladder cancer developing in a Japanese man with cerebrotendinous xanthomatosis: genetic analysis of the sterol 27-hydroxylase and p53 genes.
MedLine Citation:
PMID:  12745462     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Cerebrotendinous xanthomatosis (CTX) is a rare autosomal recessive lipid-storage disorder characterised by xanthomas, neurological dysfunctions and premature atherosclerosis. A case of a well differentiated adenocarcinoma of the gallbladder occurring in a 57-year-old Japanese man with CTX, confirmed clinically, biochemically and at autopsy is reported together with analyses of the sterol 27-hydroxylase (CYP27) and p53 genes. A missense mutation of the p53 (G for C) was detected in the gallbladder adenocarcinoma. Direct sequence analysis also showed a silent mutational substitution of unknown significance, C for A, in CYP27 at codon 89. In the past, CTX patients have only demonstrated this infrequently, indicating no direct relationship between CYP27 dysfunction and tumour development. Thus, the present case of gallbladder cancer appears to be a chance occurrence.
Authors:
Hiroya Kato; Sukenari Koyabu; Shigenori Aoki; Takuya Tamai; Masahiro Sugawa; Masatoshi Watanabe; Taizo Shiraishi
Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Pathology     Volume:  35     ISSN:  0031-3025     ISO Abbreviation:  Pathology     Publication Date:  2003 Apr 
Date Detail:
Created Date:  2003-05-14     Completed Date:  2003-07-29     Revised Date:  2012-01-30    
Medline Journal Info:
Nlm Unique ID:  0175411     Medline TA:  Pathology     Country:  England    
Other Details:
Languages:  eng     Pagination:  141-4     Citation Subset:  IM    
Affiliation:
Department of Internal Medicine, Owase General Hospital, Owase, Mie, Japan. kato2001@clin.medic.mie-u.ac.jp
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MeSH Terms
Descriptor/Qualifier:
Achilles Tendon / metabolism,  pathology
Adenocarcinoma / complications,  genetics*,  metabolism,  pathology*
Cytochrome P-450 CYP27A1
DNA Mutational Analysis
DNA, Neoplasm / analysis
Gallbladder Neoplasms / complications,  genetics*,  metabolism,  pathology*
Genes, p53*
Humans
Immunoenzyme Techniques
Male
Middle Aged
Mutation, Missense
Steroid Hydroxylases / genetics*,  metabolism
Tumor Markers, Biological / metabolism
Tumor Suppressor Protein p53 / genetics,  metabolism
Xanthomatosis, Cerebrotendinous / complications,  genetics*,  metabolism,  pathology*
Chemical
Reg. No./Substance:
0/DNA, Neoplasm; 0/Tumor Markers, Biological; 0/Tumor Suppressor Protein p53; EC 1.14.-/Cytochrome P-450 CYP27A1; EC 1.14.-/Steroid Hydroxylases; EC 1.14.13.15/CYP27A1 protein, human

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


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