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PMID: 3353383 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

Genetic heterogeneity in type 1 Gaucher disease: multiple genotypes in Ashkenazic and non-Ashkenazic individuals.

Tsuji S, Martin BM, Barranger JA, Stubblefield BK, LaMarca ME, Ginns EI

Abstract

Nucleotide sequence analysis of a genomic clone from an Ashkenazic Jewish patient with type 1 Gaucher disease revealed a single-base mutation (adenosine to guanosine transition) in exon 9 of the glucocerebrosidase gene. This change results in the amino acid substitution of serine for asparagine. Transient expression studies following oligonucleotide-directed mutagenesis of the normal cDNA confirmed that the mutation results in loss of glucocerebrosidase activity. Allele-specific hybridization with oligonucleotide probes demonstrated that this mutation was found exclusively in the type 1 phenotype. None of the 6 type 2 patients, 11 type 3 patients, or 12 normal controls had this allele. In contrast, 15 of 24 type 1 patients had one allele with this mutation, and 3 others were homozygous for the mutation. Furthermore, some of the Ashkenazic Jewish type 1 patients had only one allele with this mutation, suggesting that even in this population there is allelic heterozygosity. These findings indicate that there are multiple allelic mutations responsible for type 1 Gaucher disease in both the Jewish and non-Jewish populations. Allelic-specific hybridization demonstrating this mutation in exon 9, used in conjunction with the Nci I restriction fragment length polymorphism described as a marker for neuronopathic Gaucher disease, provides a tool for diagnosis and genetic counseling that is approximately equal to 80% informative in all Gaucher patients studied.

MeSH Terms
Adolescent Adult Alleles Amino Acid Sequence Base Sequence Child Child, Preschool Gaucher Disease/classification,enzymology,genetics Genotype Glucosidases/genetics Glucosylceramidase/deficiency,genetics Humans Infant Jews Middle Aged Molecular Sequence Data Mutation Risk
Chemicals
Glucosidases Glucosylceramidase
Authors & Affiliations
6 authors, click to expand affiliations / ORCID
Tsuji S
Molecular Neurogenetics Unit, National Institute of Mental Health, ADAMHA, Bethesda, MD 20892.
Martin B M
Barranger J A
Stubblefield B K
LaMarca M E
Ginns E I
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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
1988-04-00
Pages
2349-52
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC279989
Subset
IM
Databases
GENBANK
M20282
Corrections
ErratumIn
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