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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