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PMID: 1683708 Published · ppublish English Case Reports Journal Article

Transmissible familial Creutzfeldt-Jakob disease associated with five, seven, and eight extra octapeptide coding repeats in the PRNP gene.

Goldfarb LG, Brown P, McCombie WR, Goldgaber D, Swergold GD, Wills PR, Cervenakova L, Baron H, Gibbs CJ, Gajdusek DC

Abstract

The PRNP gene, encoding the amyloid precursor protein that is centrally involved in Creutzfeldt-Jakob disease (CJD), has an unstable region of five variant tandem octapeptide coding repeats between codons 51 and 91. We screened a total of 535 individuals for the presence of extra repeats in this region, including patients with sporadic and familial forms of spongiform encephalopathy, members of their families, other neurological and non-neurological patients, and normal controls. We identified three CJD families (in each of which the proband's disease was neuropathologically confirmed and experimentally transmitted to primates) that were heterozygous for alleles with 10, 12, or 13 repeats, some of which had "wobble" nucleotide substitutions. We also found one individual with 9 repeats and no nucleotide substitutions who had no evidence of neurological disease. These observations, together with data on published British patients with 11 and 14 repeats, strongly suggest that the occurrence of 10 or more octapeptide repeats in the encoded amyloid precursor protein predisposes to CJD.

MeSH Terms
Adult Alleles Amino Acid Sequence Amyloid beta-Protein Precursor/genetics Animals Base Sequence Brain/pathology Cloning, Molecular/methods Creutzfeldt-Jakob Syndrome/genetics,physiopathology,transmission Crossing Over, Genetic Female Humans Male Middle Aged Molecular Sequence Data Mutation Oligodeoxyribonucleotides Phenotype Polymerase Chain Reaction/methods PrPSc Proteins Primates Prions/genetics Repetitive Sequences, Nucleic Acid
Chemicals
Amyloid beta-Protein Precursor Oligodeoxyribonucleotides PrPSc Proteins Prions
Authors & Affiliations
10 authors, click to expand affiliations / ORCID
Goldfarb L G
Laboratory of Central Nervous System Studies, National Institute of Neurological Disorders and Stroke, National Institutes of Health, Bethesda, MD 20892.
Brown P
McCombie W R
Goldgaber D
Swergold G D
Wills P R
Cervenakova L
Baron H
Gibbs C J
Gajdusek D C
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Article Info
Journal
Proceedings of the National Academy of Sciences of the United States of America
Abbr.
Proc Natl Acad Sci U S A
ISSN
0027-8424
Published
1991-12-01
Pages
10926-30
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC53045
Subset
IM
Databases
GENBANK
M80723, M80724, M81661, M81662, M81664, M81665, M81666, S71208, S71210, S71212
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