Literature DB >> 2269438

Prevalent and rare mutations among Gaucher patients.

N Eyal1, S Wilder, M Horowitz.   

Abstract

Sequence analysis of gcs cDNA (encoding glucocerebrosidase) or genomic fragments originated from Gaucher patients revealed novel mutations. Two rare mutations were found in a type-2 non-Jewish Gaucher patient: a G----A transition (Gly325----Arg) at nucleotide (nt) 5306 of the active gene and a T----G transversion (Cys342----Gly) at nt 5357. These mutations were not found in any other patient. A G----C transversion (Asp409----His) at nt 5957 was identified in two non-Jewish patients, and was designated TL. Two recombinant alleles were found. One recombinant allele designated recTL contained four single-nt mutations. These mutations included: (1) a G----C transversion at nt 5957 (Asp409----His) (the TL mutation); (2) a T----C transition at nt 6433 (Leu444----Pro) creating a new NciI site (NciI mutation); (3) a G----C transversion at nt 6468 (Ala456----Pro; 456 mutation); and (4) a G----C transversion at amino acid (aa) 460 (nt 6482), not associated with any aa change. Sequence analysis indicated that at least part of exon 9, intron 9 and exon 10 of the recombinant gene derived from the pseudogene. The other recombinant gene, designated recNciI, contained a mutation at aa 444 (NciI mutation), and mutations 456 and 460 described above; at least exon 10 of this gene originated from the pseudogene. We hypothesize that the presence of the pseudogene close to the active gene causes transfer of mutations into the active gene via gene conversion or nonhomologous recombination, thus accounting for the high frequency of mutations observed in the gcs gene.

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Year:  1990        PMID: 2269438     DOI: 10.1016/0378-1119(90)90264-r

Source DB:  PubMed          Journal:  Gene        ISSN: 0378-1119            Impact factor:   3.688


  23 in total

1.  Ex vivo and in vivo effects of isofagomine on acid β-glucosidase variants and substrate levels in Gaucher disease.

Authors:  Ying Sun; Benjamin Liou; You-Hai Xu; Brian Quinn; Wujuan Zhang; Rick Hamler; Kenneth D R Setchell; Gregory A Grabowski
Journal:  J Biol Chem       Date:  2011-12-13       Impact factor: 5.157

2.  Gaucher disease in sheep.

Authors:  Litsa Karageorgos; Malcolm J Lancaster; Judith S Nimmo; John J Hopwood
Journal:  J Inherit Metab Dis       Date:  2010-10-27       Impact factor: 4.982

3.  Intracellular degradation of fluorescent glycolipids by lysosomal enzymes and their activators.

Authors:  L Madar-Shapiro; M Pasmanik-Chor; T Dinur; A Dagan; S Gatt; M Horowitz
Journal:  J Inherit Metab Dis       Date:  1999-06       Impact factor: 4.982

Review 4.  Gaucher disease as a paradigm of current issues regarding single gene mutations of humans.

Authors:  E Beutler
Journal:  Proc Natl Acad Sci U S A       Date:  1993-06-15       Impact factor: 11.205

Review 5.  The glucocerebrosidase locus in Gaucher's disease: molecular analysis of a lysosomal enzyme.

Authors:  P K Mistry; T M Cox
Journal:  J Med Genet       Date:  1993-11       Impact factor: 6.318

6.  Prevalence of nine mutations among Jewish and non-Jewish Gaucher disease patients.

Authors:  M Horowitz; G Tzuri; N Eyal; A Berebi; E H Kolodny; R O Brady; N W Barton; A Abrahamov; A Zimran
Journal:  Am J Hum Genet       Date:  1993-10       Impact factor: 11.025

7.  Phenotypic and genotypic heterogeneity in gaucher disease: Implications for genetic counseling.

Authors:  E Sidransky; E I Ginns
Journal:  J Genet Couns       Date:  1994-03       Impact factor: 2.537

8.  Three unique base pair changes in a family with Gaucher disease.

Authors:  N Eyal; N Firon; S Wilder; E H Kolodny; M Horowitz
Journal:  Hum Genet       Date:  1991-07       Impact factor: 4.132

9.  Gene conversion causing human inherited disease: evidence for involvement of non-B-DNA-forming sequences and recombination-promoting motifs in DNA breakage and repair.

Authors:  Nadia Chuzhanova; Jian-Min Chen; Albino Bacolla; George P Patrinos; Claude Férec; Robert D Wells; David N Cooper
Journal:  Hum Mutat       Date:  2009-08       Impact factor: 4.878

10.  In vivo and ex vivo evaluation of L-type calcium channel blockers on acid beta-glucosidase in Gaucher disease mouse models.

Authors:  Ying Sun; Benjamin Liou; Brian Quinn; Huimin Ran; You-Hai Xu; Gregory A Grabowski
Journal:  PLoS One       Date:  2009-10-07       Impact factor: 3.240

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