Literature DB >> 2278539

Ashkenazi-Jewish and non-Jewish adult GM2 gangliosidosis patients share a common genetic defect.

R Navon1, E H Kolodny, H Mitsumoto, G H Thomas, R L Proia.   

Abstract

The adult form of Tay-Sachs disease, adult GM2 gangliosidosis, is an autosomal recessive neurological disorder caused by a partial deficiency of beta-hexosaminidase A. We had previously identified, in Ashkenazi-Jewish adult GM2 gangliosidosis patients, a Gly269----Ser mutation in the beta-hexosaminidase alpha-subunit. All of the Ashkenazi patients were found to be compound heterozygotes with an allele containing the Gly269----Ser mutation together with one of the Ashkenazi infantile Tay-Sachs alleles. We have now found the same Gly269----Ser mutation in six adult GM2 gangliosidosis patients from four different non-Jewish families. Genomic DNA from three of the patients, two of whom were brothers, exhibited a hybridization pattern consistent with homozygosity for the Gly269----Ser mutation. The remaining non-Jewish patients were compound heterozygotes of the Gly269----Ser mutation together with an unidentified alpha-subunit mutation. The results demonstrate that individuals homozygous for the Gly269----Ser change can be clinically affected. The same Gly269----Ser mutation in both the Ashkenazi and non-Jewish patients may be the result of a common ancestor, given that the ancestry of these non-Jewish patients, like the Ashkenazim, can be traced to eastern Europe.

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Year:  1990        PMID: 2278539      PMCID: PMC1683663     

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


  14 in total

Review 1.  Natural history and inherited disorders of a lysosomal enzyme, beta-hexosaminidase.

Authors:  E F Neufeld
Journal:  J Biol Chem       Date:  1989-07-05       Impact factor: 5.157

2.  Identification of an altered splice site in Ashkenazi Tay-Sachs disease.

Authors:  E Arpaia; A Dumbrille-Ross; T Maler; K Neote; M Tropak; C Troxel; J L Stirling; J S Pitts; B Bapat; A M Lamhonwah
Journal:  Nature       Date:  1988-05-05       Impact factor: 49.962

3.  Primer-directed enzymatic amplification of DNA with a thermostable DNA polymerase.

Authors:  R K Saiki; D H Gelfand; S Stoffel; S J Scharf; R Higuchi; G T Horn; K B Mullis; H A Erlich
Journal:  Science       Date:  1988-01-29       Impact factor: 47.728

4.  Frequency of hexosaminidase A variant alleles among Ashkenazi Jews and prenatal diagnosis of GM2 gangliosidosis.

Authors:  R Navon; A Adam
Journal:  Am J Hum Genet       Date:  1985-09       Impact factor: 11.025

5.  Ganglioside GM2 N-acetyl-beta-D-galactosaminidase activity in cultured fibroblasts of late-infantile and adult GM2 gangliosidosis patients and of healthy probands with low hexosaminidase level.

Authors:  E Conzelmann; H J Kytzia; R Navon; K Sandhoff
Journal:  Am J Hum Genet       Date:  1983-09       Impact factor: 11.025

6.  Hexosaminidase A deficiency in adults.

Authors:  R Navon; Z Argov; A Frisch
Journal:  Am J Med Genet       Date:  1986-05

7.  Organization of the gene encoding the human beta-hexosaminidase alpha-chain.

Authors:  R L Proia; E Soravia
Journal:  J Biol Chem       Date:  1987-04-25       Impact factor: 5.157

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

Authors:  R Navon; R L Proia
Journal:  Science       Date:  1989-03-17       Impact factor: 47.728

9.  Estimation of the frequency of hexosaminidase a variant alleles in the American Jewish population.

Authors:  D A Greenberg; M M Kaback
Journal:  Am J Hum Genet       Date:  1982-05       Impact factor: 11.025

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  17 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.  A sensitive fluorescence-based assay for monitoring GM2 ganglioside hydrolysis in live patient cells and their lysates.

Authors:  Michael B Tropak; Scott W Bukovac; Brigitte A Rigat; Sayuri Yonekawa; Warren Wakarchuk; Don J Mahuran
Journal:  Glycobiology       Date:  2009-11-16       Impact factor: 4.313

3.  Hexosaminidase--pseudodeficiency?

Authors:  J Kappler; V Gieselmann; P Propping
Journal:  Am J Hum Genet       Date:  1990-11       Impact factor: 11.025

4.  Identification of a patient affected by "Juvenile-chronic" Tay Sachs disease in South Italy.

Authors:  M Liguori; G Tagarelli; N Romeo; A Bagalà; Patrizia Spadafora
Journal:  Neurol Sci       Date:  2016-06-28       Impact factor: 3.307

Review 5.  Chaperone therapy for GM2 gangliosidosis: effects of pyrimethamine on β-hexosaminidase activity in Sandhoff fibroblasts.

Authors:  Elena Chiricozzi; Natalia Niemir; Massimo Aureli; Alessandro Magini; Nicoletta Loberto; Alessandro Prinetti; Rosaria Bassi; Alice Polchi; Carla Emiliani; Catherine Caillaud; Sandro Sonnino
Journal:  Mol Neurobiol       Date:  2013-12-20       Impact factor: 5.590

6.  Frequency of three Hex A mutant alleles among Jewish and non-Jewish carriers identified in a Tay-Sachs screening program.

Authors:  B H Paw; P T Tieu; M M Kaback; J Lim; E F Neufeld
Journal:  Am J Hum Genet       Date:  1990-10       Impact factor: 11.025

7.  Molecular genetics of Tay-Sachs disease in Japan.

Authors:  A Tanaka; H Sakazaki; H Murakami; G Isshiki; K Suzuki
Journal:  J Inherit Metab Dis       Date:  1994       Impact factor: 4.982

Review 8.  Frontotemporal dementia: a bridge between dementia and neuromuscular disease.

Authors:  Adeline S L Ng; Rosa Rademakers; Bruce L Miller
Journal:  Ann N Y Acad Sci       Date:  2014-12-30       Impact factor: 5.691

Review 9.  Lending a helping hand, screening chemical libraries for compounds that enhance beta-hexosaminidase A activity in GM2 gangliosidosis cells.

Authors:  Michael B Tropak; Don Mahuran
Journal:  FEBS J       Date:  2007-10       Impact factor: 5.542

10.  Six novel deleterious and three neutral mutations in the gene encoding the alpha-subunit of hexosaminidase A in non-Jewish individuals.

Authors:  E H Mules; S Hayflick; C S Miller; L W Reynolds; G H Thomas
Journal:  Am J Hum Genet       Date:  1992-04       Impact factor: 11.025

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