Literature DB >> 1532289

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

E H Mules1, S Hayflick, C S Miller, L W Reynolds, G H Thomas.   

Abstract

Initial investigations demonstrated that only 3/34 "Tay-Sachs chromosomes" in 22 unrelated, non-Jewish patients or carriers of some form of GM2-gangliosidosis (7 black and 15 non-Jewish Caucasian) had either of the two mutations commonly found in the Jewish population. To determine the nature and incidence of the alterations in this non-Jewish population we have utilized PCR, single-strand conformation polymorphism analysis and sequencing to detect new mutations in genomic DNA. Fourteen primer sets have been utilized to analyze 80% of the coding region and 23/26 splice sites of the gene coding for the alpha chain of hexosaminidase A. Presumed deleterious mutations were discovered in 17/34 chromosomes believed to be carrying a beta-hexosaminidase A alpha-subunit gene mutation. Ten had abnormalities which have been described previously. In the remaining 24 Tay-Sachs disease alleles, six novel mutations predicted to be deleterious were discovered. These include two small deletions (a single-base frameshift and a three-base deletion removing an amino acid), two different nonsense mutations, an initiation codon mutation (ATG----GTG), and a missense mutation (Arg499Cys) in a highly conserved residue. In addition, three presumed nondeleterious mutations were found.

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Year:  1992        PMID: 1532289      PMCID: PMC1682641     

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


  29 in total

1.  A frameshift mutation in a patient with Tay-Sachs disease causes premature termination and defective intracellular transport of the alpha-subunit of beta-hexosaminidase.

Authors:  M M Lau; E F Neufeld
Journal:  J Biol Chem       Date:  1989-12-15       Impact factor: 5.157

2.  Screening for carriers of Tay-Sachs disease among Ashkenazi Jews. A comparison of DNA-based and enzyme-based tests.

Authors:  B L Triggs-Raine; A S Feigenbaum; M Natowicz; M A Skomorowski; S M Schuster; J T Clarke; D J Mahuran; E H Kolodny; R A Gravel
Journal:  N Engl J Med       Date:  1990-07-05       Impact factor: 91.245

3.  Functionally important regions of the factor IX gene have a low rate of polymorphism and a high rate of mutation in the dinucleotide CpG.

Authors:  D D Koeberl; C D Bottema; J M Buerstedde; S S Sommer
Journal:  Am J Hum Genet       Date:  1989-09       Impact factor: 11.025

4.  A point mutation in the coding sequence of the beta-hexosaminidase alpha gene results in defective processing of the enzyme protein in an unusual GM2-gangliosidosis variant.

Authors:  T Nakano; M Muscillo; K Ohno; A J Hoffman; K Suzuki
Journal:  J Neurochem       Date:  1988-09       Impact factor: 5.372

5.  GM2-gangliosidosis B1 variant: analysis of beta-hexosaminidase alpha gene abnormalities in seven patients.

Authors:  A Tanaka; K Ohno; K Sandhoff; I Maire; E H Kolodny; A Brown; K Suzuki
Journal:  Am J Hum Genet       Date:  1990-02       Impact factor: 11.025

6.  The major defect in Ashkenazi Jews with Tay-Sachs disease is an insertion in the gene for the alpha-chain of beta-hexosaminidase.

Authors:  R Myerowitz; F C Costigan
Journal:  J Biol Chem       Date:  1988-12-15       Impact factor: 5.157

7.  A new point mutation within exon 5 of beta-hexosaminidase alpha gene in a Japanese infant with Tay-Sachs disease.

Authors:  T Nakano; E Nanba; A Tanaka; K Ohno; Y Suzuki; K Suzuki
Journal:  Ann Neurol       Date:  1990-05       Impact factor: 10.422

8.  Late onset GM2 gangliosidosis: an alpha-locus genetic compound with near normal hexosaminidase activity.

Authors:  J Charrow; K Inui; D A Wenger
Journal:  Clin Genet       Date:  1985-01       Impact factor: 4.438

9.  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

10.  Molecular cloning of the cDNA which encodes beta-N-acetylhexosaminidase A from Dictyostelium discoideum. Complete amino acid sequence and homology with the human enzyme.

Authors:  T R Graham; H P Zassenhaus; A Kaplan
Journal:  J Biol Chem       Date:  1988-11-15       Impact factor: 5.157

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  11 in total

1.  Structural basis of the GM2 gangliosidosis B variant.

Authors:  Fumiko Matsuzawa; Sei-ichi Aikawa; Hitoshi Sakuraba; Hoang Thi Ngoc Lan; Akemi Tanaka; Kousaku Ohno; Yuko Sugimoto; Haruaki Ninomiya; Hirofumi Doi
Journal:  J Hum Genet       Date:  2003-10-24       Impact factor: 3.172

2.  Tay-Sachs disease preconception screening in Australia: self-knowledge of being an Ashkenazi Jew predicts carrier state better than does ancestral origin, although there is an increased risk for c.1421 + 1G > C mutation in individuals with South African heritage.

Authors:  Raelia Lew; Leslie Burnett; Anné Proos
Journal:  J Community Genet       Date:  2011-07-15

3.  Novel mutations and DNA-based screening in non-Jewish carriers of Tay-Sachs disease.

Authors:  B R Akerman; M R Natowicz; M M Kaback; M Loyer; E Campeau; R A Gravel
Journal:  Am J Hum Genet       Date:  1997-05       Impact factor: 11.025

4.  Further investigation of the HEXA gene intron 9 donor splice site mutation frequently found in non-Jewish Tay-Sachs disease patients from the British Isles.

Authors:  E C Landels; P M Green; I H Ellis; A H Fensom; M M Kaback; J Lim-Steele; K Zeiger; N Levy; M Bobrow
Journal:  J Med Genet       Date:  1993-06       Impact factor: 6.318

5.  Different attenuated phenotypes of GM2 gangliosidosis variant B in Japanese patients with HEXA mutations at codon 499, and five novel mutations responsible for infantile acute form.

Authors:  Akemi Tanaka; Lan Thi Ngcok Hoang; Yasuaki Nishi; Satoshi Maniwa; Makio Oka; Tsunekazu Yamano
Journal:  J Hum Genet       Date:  2003-10-18       Impact factor: 3.172

6.  Mutational analyses of Tay-Sachs disease: studies on Tay-Sachs carriers of French Canadian background living in New England.

Authors:  B Triggs-Raine; M Richard; N Wasel; E M Prence; M R Natowicz
Journal:  Am J Hum Genet       Date:  1995-04       Impact factor: 11.025

7.  Identification and rapid detection of three Tay-Sachs mutations in the Moroccan Jewish population.

Authors:  L Drucker; R L Proia; R Navon
Journal:  Am J Hum Genet       Date:  1992-08       Impact factor: 11.025

8.  Two new mutations in a late infantile Tay-Sachs patient are both in exon 1 of the beta-hexosaminidase alpha subunit gene.

Authors:  D L Harmon; D Gardner-Medwin; J L Stirling
Journal:  J Med Genet       Date:  1993-02       Impact factor: 6.318

9.  Identification of novel variants in a large cohort of children with Tay-Sachs disease: An initiative of a multicentric task force on lysosomal storage disorders by Government of India.

Authors:  Mehul Mistri; Sanjeev Mehta; Dhaval Solanki; Mahesh Kamate; Neerja Gupta; Madhulika Kabra; Ratna Puri; Katta Girisha; Sankar Hariharan; Sheela Nampoothiri; Frenny Sheth; Jayesh Sheth
Journal:  J Hum Genet       Date:  2019-08-06       Impact factor: 3.172

10.  Tay-Sachs Carrier Screening by Enzyme and Molecular Analyses in the New York City Minority Population.

Authors:  Nikita Mehta; Gabriel A Lazarin; Erica Spiegel; Kathleen Berentsen; Kelly Brennan; Jessica Giordano; Imran S Haque; Ronald Wapner
Journal:  Genet Test Mol Biomarkers       Date:  2016-06-30
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