Document Detail


Influence of homozygosity for methionine at codon 129 of the human prion gene on the onset of neurological and hepatic symptoms in Wilson disease.
MedLine Citation:
PMID:  16831968     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
BACKGROUND: The clinical heterogeneity of Wilson disease expression cannot be fully explained by the various mutations of the Wilson disease gene. The prion-related protein (PrP) has been shown to bind copper in vitro and might therefore influence Wilson disease. OBJECTIVE: To examine the effect of the PrP polymorphism at codon 129, resulting in either methionine or valine (M129V), on the clinical phenotype of patients with Wilson disease. DESIGN AND SETTING: Retrospective cross-sectional study at a university hospital. PARTICIPANTS: A total of 134 patients were grouped according to their PrP M129V genotypes and initial clinical symptoms (hepatic vs neurological). RESULTS: The onset of symptoms was significantly delayed in patients homozygous for the 129M allele as compared with patients with at least 1 V allele (mean +/- SD age, 20.90 +/- 11.9 years vs 15.5 +/- 7.6 years; P = .003). No significant correlation was found when analyzing the impact of the PrP M129V genotype on the clinical symptoms at initial manifestation (hepatic vs neurological; P = .44). CONCLUSION: This study shows for the first time, to our knowledge, that the human PrP polymorphism M129V influences the onset of symptoms in patients with the copper storage disorder Wilson disease.
Authors:
Uta Merle; Wolfgang Stremmel; Reinhard Gessner
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Archives of neurology     Volume:  63     ISSN:  0003-9942     ISO Abbreviation:  Arch. Neurol.     Publication Date:  2006 Jul 
Date Detail:
Created Date:  2006-07-11     Completed Date:  2006-08-07     Revised Date:  2006-11-15    
Medline Journal Info:
Nlm Unique ID:  0372436     Medline TA:  Arch Neurol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  982-5     Citation Subset:  AIM; IM    
Affiliation:
Department of Gastroenterology and Hepatology, University Hospital, Heidelberg, Germany. uta_merle@med.uni-heidelberg.de
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Adult
Age of Onset
Apolipoprotein E3
Apolipoproteins E / genetics
Codon
Copper / metabolism
Cross-Sectional Studies
Female
Genotype
Hepatolenticular Degeneration / complications,  genetics*,  metabolism
Homozygote
Humans
Liver Diseases / etiology,  genetics,  metabolism
Male
Methionine / genetics
Nervous System Diseases / etiology,  genetics,  metabolism
Polymorphism, Genetic
Prions / genetics*,  metabolism
Retrospective Studies
Chemical
Reg. No./Substance:
0/Apolipoprotein E3; 0/Apolipoproteins E; 0/Codon; 0/Prions; 63-68-3/Methionine; 7440-50-8/Copper

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


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