Literature DB >> 808963

Hexosaminidase isozyme in type O Gm2 gangliosidosis (Sandhoff-Jatzkewitz disease).

E Beutler, W Kuhl, D Comings.   

Abstract

The residual enzyme of the fibroblasts of a child with homozygous type 0 GM2 gangliosidosis (Sandhoff-Jatzkewitz disease) has been found to correspond with a minor fraction of enzyme which can be isolated from normal fibroblasts by repeated chromatography. This enzyme is designated as hexosaminidase (hex) S. It reacts with antiserum prepared against homogeneous hex A but not with serum prepared against homogeneous hex B. These findings support our previously described model of the relationship between hex A and hex G: hex A has the structure (alpha beta)3, while hex B is (beta)6. Type B GM2 gangliosidosis (Tay-Sachs disease) is the alpha- mutation, while type 0 GM2 gangliosidosis (Sandhoff-Jatzkewitz disease) is the beta- mutation. In the absence of normal beta subunits there is increased polymerization of alpha subunits forming hex S, which probably has a structure of (alpha)6. A parallel between the thalassemias and GM2 gangliosidosis is evident: deficiency of one of the chains of which the protein is composed leads to an excess of polymers comprised of the other chains. In type B GM2 gangliosidosis, the excess of beta chanis leads to increased amounts of hex B beta)6; in type 0 GM2 gangliosidosis, the excess of alpha chains leads to formation of increased amounts of the alpha chain polymer, hex S.

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Year:  1975        PMID: 808963      PMCID: PMC1762825     

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


  22 in total

1.  Amino acid metabolism in mammalian cell cultures.

Authors:  H EAGLE
Journal:  Science       Date:  1959-08-21       Impact factor: 47.728

2.  Isolation and relationship of human hexosaminidases.

Authors:  J F Tallman; R O Brady; J M Quirk; M Villalba; A E Gal
Journal:  J Biol Chem       Date:  1974-06-10       Impact factor: 5.157

3.  Studies on human beta-D-N-acetylhexosaminidases. I. Purification and properties.

Authors:  S K Srivastava; Y C Awasthi; A Yoshida; E Beutler
Journal:  J Biol Chem       Date:  1974-04-10       Impact factor: 5.157

4.  Genetic complementation after fusion of Tay-Sachs and Sandhoff cells.

Authors:  G H Thomas; H A Taylor; C S Miller; J Axelman; B R Migeon
Journal:  Nature       Date:  1974-08-16       Impact factor: 49.962

5.  Variability of -galactosidase A and B in different tissues of man.

Authors:  E Beutler; E Guinto; W Kuhl
Journal:  Am J Hum Genet       Date:  1973-01       Impact factor: 11.025

6.  X-linkage of human -galactosidase.

Authors:  K H Grzeschik; A M Grzeschik; S Banhof; G Romeo; M Siniscalco; H van Someren; P Meera Khan; A Westerveld; D Bootsma
Journal:  Nat New Biol       Date:  1972-11-08

7.  Immunological identity of human liver hexosaminidases.

Authors:  M Carroll; D Robinson
Journal:  Biochem J       Date:  1972-02       Impact factor: 3.857

8.  Studies on human beta-D-N-acetylhexosaminidases. 3. Biochemical genetics of Tay-Sachs and Sandhoff's diseases.

Authors:  S K Srivastava; E Beutler
Journal:  J Biol Chem       Date:  1974-04-10       Impact factor: 5.157

9.  Human beta-D-N-acetylhexosaminidases A and B: expression and linkage relationships in somatic cell hybrids.

Authors:  P A Lalley; M C Rattazzi; T B Shows
Journal:  Proc Natl Acad Sci U S A       Date:  1974-04       Impact factor: 11.205

10.  Tay-Sachs' and Sandhoff's diseases: the assignment of genes for hexosaminidase A and B to individual human chromosomes.

Authors:  F Gilbert; R Kucherlapati; R P Creagan; M J Murnane; G J Darlington; F H Ruddle
Journal:  Proc Natl Acad Sci U S A       Date:  1975-01       Impact factor: 11.205

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  30 in total

Review 1.  Biochemistry and genetics of gangliosidoses.

Authors:  K Sandhoff; H Christomanou
Journal:  Hum Genet       Date:  1979       Impact factor: 4.132

Review 2.  Basic findings and current developments in sphingolipidoses.

Authors:  H Pilz; R Heipertz; D Seidel
Journal:  Hum Genet       Date:  1979-03-12       Impact factor: 4.132

3.  The subunits of human hexosaminidase A.

Authors:  E Beutler; A Yoshida; W Kuhl; J E Lee
Journal:  Biochem J       Date:  1976-12-01       Impact factor: 3.857

Review 4.  Glycosphingolipid hydrolases: properties and molecular genetics.

Authors:  M Wan Ho; A G Norden; J A Alhadeff; J S O'Brien
Journal:  Mol Cell Biochem       Date:  1977-10-07       Impact factor: 3.396

Review 5.  Progress in investigations of sphingolipidoses.

Authors:  M Adachi; L Schneck; B W Volk
Journal:  Acta Neuropathol       Date:  1978-08-07       Impact factor: 17.088

6.  Characterization of proteins structurally related to human N-acetyl-beta-D-glucosaminidase.

Authors:  M Carroll
Journal:  Biochem J       Date:  1978-07-01       Impact factor: 3.857

7.  Studies on human N-acetyl-Beta-d-hexosaminidase C separated from neonatal brain.

Authors:  G T Besley; D M Broadhead
Journal:  Biochem J       Date:  1976-04-01       Impact factor: 3.857

8.  Clinical,biochemical and molecular analysis of five Chinese patients with Sandhoff disease.

Authors:  Wen Zhang; Huasong Zeng; Yonglan Huang; Ting Xie; Jipeng Zheng; Xiaoyuan Zhao; Huiying Sheng; Hongsheng Liu; Li Liu
Journal:  Metab Brain Dis       Date:  2016-03-28       Impact factor: 3.584

9.  Incidence and carrier frequency of Sandhoff disease in Saskatchewan determined using a novel substrate with detection by tandem mass spectrometry and molecular genetic analysis.

Authors:  Braden Fitterer; Patricia Hall; Nick Antonishyn; Rajagopal Desikan; Michael Gelb; Denis Lehotay
Journal:  Mol Genet Metab       Date:  2014-01-13       Impact factor: 4.797

10.  Thermal activation of hexosaminidase A in a genetic compound with Tay-Sachs disease.

Authors:  Y Ben-Yoseph; M S Baylerian; T Momoi; H L Nadler
Journal:  J Inherit Metab Dis       Date:  1983       Impact factor: 4.982

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