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PMID: 10777718 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Gaucher disease: the origins of the Ashkenazi Jewish N370S and 84GG acid beta-glucosidase mutations.

American journal of human genetics ·Vol. 66 ·No. 6 ·2000-06-00 ·Pages 1821-32

Diaz GA, Gelb BD, Risch N, Nygaard TG, Frisch A, Cohen IJ, Miranda CS, Amaral O, Maire I, Poenaru L, Caillaud C, Weizberg M, Mistry P, Desnick RJ

Abstract

Type 1 Gaucher disease (GD), a non-neuronopathic lysosomal storage disorder, results from the deficient activity of acid beta-glucosidase (GBA). Type 1 disease is panethnic but is more prevalent in individuals of Ashkenazi Jewish (AJ) descent. Of the causative GBA mutations, N370S is particularly frequent in the AJ population, (q approximately .03), whereas the 84GG insertion (q approximately .003) occurs exclusively in the Ashkenazim. To investigate the genetic history of these mutations in the AJ population, short tandem repeat (STR) markers were used to map a 9.3-cM region containing the GBA locus and to genotype 261 AJ N370S chromosomes, 60 European non-Jewish N370S chromosomes, and 62 AJ 84GG chromosomes. A highly conserved haplotype at four markers flanking GBA (PKLR, D1S1595, D1S2721, and D1S2777) was observed on both the AJ chromosomes and the non-Jewish N370S chromosomes, suggesting the occurrence of a founder common to both populations. Of note, the presence of different divergent haplotypes suggested the occurrence of de novo, recurrent N370S mutations. In contrast, a different conserved haplotype at these markers was identified on the 84GG chromosomes, which was unique to the AJ population. On the basis of the linkage disequilibrium (LD) delta values, the non-Jewish European N370S chromosomes had greater haplotype diversity and less LD at the markers flanking the conserved haplotype than did the AJ N370S chromosomes. This finding is consistent with the presence of the N370S mutation in the non-Jewish European population prior to the founding of the AJ population. Coalescence analyses for the N370S and 84GG mutations estimated similar coalescence times, of 48 and 55.5 generations ago, respectively. The results of these studies are consistent with a significant bottleneck occurring in the AJ population during the first millennium, when the population became established in Europe.

MeSH Terms
Algorithms Amino Acid Substitution/genetics Chromosome Mapping Conserved Sequence/genetics Europe Founder Effect Gaucher Disease/enzymology,genetics Gene Frequency/genetics Genetic Markers/genetics Glucosylceramidase/genetics Haplotypes/genetics Humans Jews/genetics Linkage Disequilibrium/genetics Mutation, Missense/genetics Tandem Repeat Sequences/genetics Time Factors
Chemicals
Genetic Markers Glucosylceramidase
Authors & Affiliations
14 authors, click to expand affiliations / ORCID
Diaz G A
Department of Human Genetics, Mount Sinai School of Medicine, New York, NY 10029, USA.
Gelb B D
Risch N
Nygaard T G
Frisch A
Cohen I J
Miranda C S
Amaral O
Maire I
Poenaru L
Caillaud C
Weizberg M
Mistry P
Desnick R J
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Article Info
Journal
American journal of human genetics
Abbr.
Am J Hum Genet
ISSN
0002-9297
Published
2000-06-00
Epub
2000-00-21
Pages
1821-32
Language
English
Region
United States
NLM ID
0370475
PMCID
PMC1378046
Subset
IM
Grants
NICHD NIH HHS · 5 P30 HD 28822 · United States
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