Literature DB >> 2522679

The mutations in Ashkenazi Jews with adult GM2 gangliosidosis, the adult form of Tay-Sachs disease.

R Navon1, R L Proia.   

Abstract

The adult form of Tay-Sachs disease, adult GM2 gangliosidosis, is an autosomal recessive disorder that results from mutations in the alpha chain of beta-hexosaminidase A. This disorder, like infantile Tay-Sachs disease, is more frequent in the Ashkenazi Jewish population. A point mutation in the alpha-chain gene was identified that results in the substitution of Gly with Ser in eight Ashkenazi adult GM2 gangliosidosis patients from five different families. This amino acid substitution was shown to depress drastically the catalytic activity of the alpha chain after expression in COS-1 cells. All of these patients proved to be compound heterozygotes of the allele with the Gly to Ser change and one of the two Ashkenazi infantile Tay-Sachs alleles. These findings will aid in the diagnosis and understanding of beta-hexosaminidase A deficiency disorders.

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Year:  1989        PMID: 2522679     DOI: 10.1126/science.2522679

Source DB:  PubMed          Journal:  Science        ISSN: 0036-8075            Impact factor:   47.728


  39 in total

1.  Cloning and sequence analysis of a cDNA encoding the alpha-subunit of mouse beta-N-acetylhexosaminidase and comparison with the human enzyme.

Authors:  T Beccari; J Hoade; A Orlacchio; J L Stirling
Journal:  Biochem J       Date:  1992-07-15       Impact factor: 3.857

2.  Detection of single base differences using biotinylated nucleotides with very long linker arms.

Authors:  K J Livak; F W Hobbs; R J Zagursky
Journal:  Nucleic Acids Res       Date:  1992-09-25       Impact factor: 16.971

3.  A double mutation in exon 6 of the beta-hexosaminidase alpha subunit in a patient with the B1 variant of Tay-Sachs disease.

Authors:  P J Ainsworth; M B Coulter-Mackie
Journal:  Am J Hum Genet       Date:  1992-10       Impact factor: 11.025

4.  Factor XI (plasma thromboplastin antecedent) deficiency in Ashkenazi Jews is a bleeding disorder that can result from three types of point mutations.

Authors:  R Asakai; D W Chung; O D Ratnoff; E W Davie
Journal:  Proc Natl Acad Sci U S A       Date:  1989-10       Impact factor: 11.205

5.  Frequency of the Tay-Sachs disease splice and insertion mutations in the UK Ashkenazi Jewish population.

Authors:  E C Landels; I H Ellis; A H Fensom; P M Green; M Bobrow
Journal:  J Med Genet       Date:  1991-03       Impact factor: 6.318

6.  Distribution of three alpha-chain beta-hexosaminidase A mutations among Tay-Sachs carriers.

Authors:  E E Grebner; J Tomczak
Journal:  Am J Hum Genet       Date:  1991-03       Impact factor: 11.025

7.  Enzyme studies in GM2 gangliosidiosis, and their application in prenatal diagnosis.

Authors:  M Kaur; I C Verma
Journal:  Indian J Pediatr       Date:  1995 Jul-Aug       Impact factor: 1.967

8.  Heterozygosity for Tay-Sachs disease in non-Jewish Americans with ancestry from Ireland or Great Britain.

Authors:  M van Bael; M R Natowicz; J Tomczak; E E Grebner; E M Prence
Journal:  J Med Genet       Date:  1996-10       Impact factor: 6.318

9.  Four novel PEPD alleles causing prolidase deficiency.

Authors:  P Ledoux; C Scriver; P Hechtman
Journal:  Am J Hum Genet       Date:  1994-06       Impact factor: 11.025

10.  beta-Hexosaminidase isozymes from cells cotransfected with alpha and beta cDNA constructs: analysis of the alpha-subunit missense mutation associated with the adult form of Tay-Sachs disease.

Authors:  C A Brown; D J Mahuran
Journal:  Am J Hum Genet       Date:  1993-08       Impact factor: 11.025

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