Literature DB >> 2464926

Characterization of mutations in Gaucher patients by cDNA cloning.

M Wigderson1, N Firon, Z Horowitz, S Wilder, Y Frishberg, O Reiner, M Horowitz.   

Abstract

Mutated cDNA clones containing the entire coding sequence of human glucocerebrosidase were isolated from libraries originated from Gaucher patients. Sequence analysis of a mutated cDNA derived from a type II Gaucher patient revealed a C-to-G transversion causing a substitution of an arginine for a proline at residue 415. This change creates a new cleavage site for the enzyme HhaI in the mutated cDNA. Allele-specific oligonucleotide hybridization made it possible to show that this mutation exists in the genomic DNA of the patient. From a cDNA library originated from a type I Gaucher patient, a mutated allele was cloned that contains a T-to-C transition causing a substitution of proline for leucine at residue 444 and creating a new NciI site. This mutation is identical to that described by S. Tsuji and colleagues in genomic DNA from type I, type II, and type III patients. Since the new NciI site generates RFLP, it was used to test the existence of this mutated allele in several Gaucher patients by Southern blot analysis. This allele was found in type I (Jewish and non-Jewish), type II, and type III Gaucher patients. These findings led us to conclude that the patient suffering from type II disease (denoted GM1260) carried both mutations described above. Any one of the amino acid changes described reduces the glucocerebrosidase activity as tested by transfection of COS cells with expression vectors harboring the mutated cDNAs. The base changes in the two mutated cDNAs do not affect the electrophoretic mobility of the corresponding polypeptides on an SDS polyacrylamide gel.

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Year:  1989        PMID: 2464926      PMCID: PMC1715434     

Source DB:  PubMed          Journal:  Am J Hum Genet        ISSN: 0002-9297            Impact factor:   11.025


  30 in total

1.  METABOLISM OF GLUCOCEREBROSIDES. II. EVIDENCE OF AN ENZYMATIC DEFICIENCY IN GAUCHER'S DISEASE.

Authors:  R O BRADY; J N KANFER; D SHAPIRO
Journal:  Biochem Biophys Res Commun       Date:  1965-01-18       Impact factor: 3.575

2.  Detection of specific sequences among DNA fragments separated by gel electrophoresis.

Authors:  E M Southern
Journal:  J Mol Biol       Date:  1975-11-05       Impact factor: 5.469

3.  Genetic disorders of lysosomes.

Authors:  R Hirschhorn; G Weissmann
Journal:  Prog Med Genet       Date:  1976

4.  Structural analysis of the human glucocerebrosidase genes.

Authors:  O Reiner; M Wigderson; M Horowitz
Journal:  DNA       Date:  1988-03

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

Authors:  S Tsuji; B M Martin; J A Barranger; B K Stubblefield; M E LaMarca; E I Ginns
Journal:  Proc Natl Acad Sci U S A       Date:  1988-04       Impact factor: 11.205

6.  Specific synthesis of DNA in vitro via a polymerase-catalyzed chain reaction.

Authors:  K B Mullis; F A Faloona
Journal:  Methods Enzymol       Date:  1987       Impact factor: 1.600

7.  The glycoprotein encoded by the X-linked chronic granulomatous disease locus is a component of the neutrophil cytochrome b complex.

Authors:  M C Dinauer; S H Orkin; R Brown; A J Jesaitis; C A Parkos
Journal:  Nature       Date:  1987 Jun 25-Jul 1       Impact factor: 49.962

8.  Cleavage of structural proteins during the assembly of the head of bacteriophage T4.

Authors:  U K Laemmli
Journal:  Nature       Date:  1970-08-15       Impact factor: 49.962

9.  Purification of biologically active globin messenger RNA by chromatography on oligothymidylic acid-cellulose.

Authors:  H Aviv; P Leder
Journal:  Proc Natl Acad Sci U S A       Date:  1972-06       Impact factor: 11.205

10.  The human glucocerebrosidase gene has two functional ATG initiator codons.

Authors:  J A Sorge; C West; W Kuhl; L Treger; E Beutler
Journal:  Am J Hum Genet       Date:  1987-12       Impact factor: 11.025

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  20 in total

1.  Comparative study on glucocerebrosidase in spleens from patients with Gaucher disease.

Authors:  J M Aerts; W E Donker-Koopman; S Brul; S Van Weely; M C Sa Miranda; J A Barranger; J M Tager; A W Schram
Journal:  Biochem J       Date:  1990-07-01       Impact factor: 3.857

Review 2.  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 3.  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

4.  Carrier testing for severe childhood recessive diseases by next-generation sequencing.

Authors:  Callum J Bell; Darrell L Dinwiddie; Neil A Miller; Shannon L Hateley; Elena E Ganusova; Joann Mudge; Ray J Langley; Lu Zhang; Clarence C Lee; Faye D Schilkey; Vrunda Sheth; Jimmy E Woodward; Heather E Peckham; Gary P Schroth; Ryan W Kim; Stephen F Kingsmore
Journal:  Sci Transl Med       Date:  2011-01-12       Impact factor: 17.956

5.  Comparison of RNase A, a chemical cleavage and GC-clamped denaturing gradient gel electrophoresis for the detection of mutations in exon 9 of the human acid beta-glucosidase gene.

Authors:  B D Theophilus; T Latham; G A Grabowski; F I Smith
Journal:  Nucleic Acids Res       Date:  1989-10-11       Impact factor: 16.971

6.  Gaucher disease: molecular heterogeneity and phenotype-genotype correlations.

Authors:  B Theophilus; T Latham; G A Grabowski; F I Smith
Journal:  Am J Hum Genet       Date:  1989-08       Impact factor: 11.025

7.  Complex alleles of the acid beta-glucosidase gene in Gaucher disease.

Authors:  T Latham; G A Grabowski; B D Theophilus; F I Smith
Journal:  Am J Hum Genet       Date:  1990-07       Impact factor: 11.025

8.  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

9.  Gaucher disease: heterologous expression of two alleles associated with neuronopathic phenotypes.

Authors:  M E Grace; A Berg; G S He; L Goldberg; M Horowitz; G A Grabowski
Journal:  Am J Hum Genet       Date:  1991-09       Impact factor: 11.025

10.  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

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