Literature DB >> 2939713

Two abnormalities of hexosaminidase A in clinically normal individuals.

E E Grebner, D A Mansfield, S S Raghavan, E H Kolodny, A d'Azzo, E F Neufeld, L G Jackson.   

Abstract

Two abnormalities of beta-hexosaminidase A (HEX A) activity are described. One, found in two unrelated Jewish children, was characterized by the complete absence of HEX A activity in serum, but low levels of activity in leukocytes and fibroblasts using artificial substrate. The other, found in a non-Jewish man, was characterized by uniformly low levels of HEX A activity in leukocytes, fibroblasts, and serum against artificial substrate. In all cases, the pH optimum of HEX A was normal, there was no increased lability at 37 degrees C, and no inhibitor was detected to account for the deficiency of activity. Cultured fibroblasts of these individuals were capable of synthesizing and processing alpha- and beta-subunits of HEX A and capable of cleaving GM2 ganglioside. The patients, ranging in age from 6 to 30 years, are clinically normal. They are probably genetic compounds carrying the classical Tay-Sachs gene and a differently mutated allele that imparts the anomalous phenotypic features observed.

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Year:  1986        PMID: 2939713      PMCID: PMC1684809     

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


  14 in total

Review 1.  Biochemistry and genetics of gangliosidoses.

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

2.  Segregation within a family of two mutant alleles for hexosaminidase A.

Authors:  T E Kelly; L W Reynolds; J S O'Brien
Journal:  Clin Genet       Date:  1976-05       Impact factor: 4.438

3.  Low levels of beta hexosaminidase A in healthy individuals with apparent deficiency of this enzyme.

Authors:  R Navon; B Geiger; Y B Yoseph; M C Rattazzi
Journal:  Am J Hum Genet       Date:  1976-07       Impact factor: 11.025

4.  Prenatal diagnosis of Tay-Sachs disease: studies on the reliability of hexosaminidase levels in amniotic fluid.

Authors:  E E Grebner; L G Jackson
Journal:  Am J Obstet Gynecol       Date:  1979-07-01       Impact factor: 8.661

5.  Absence of -N-acetyl-D-hexosaminidase A activity in a healthy woman.

Authors:  J Vidgoff; N R Buist; J S O'Brien
Journal:  Am J Hum Genet       Date:  1973-07       Impact factor: 11.025

6.  Biosynthesis of lysosomal enzymes in fibroblasts. Synthesis as precursors of higher molecular weight.

Authors:  A Hasilik; E F Neufeld
Journal:  J Biol Chem       Date:  1980-05-25       Impact factor: 5.157

7.  Tay-Sachs disease heterozygote detection: a quality control study.

Authors:  M M Kaback; L J Shapiro; P Hirsch; C Roy
Journal:  Prog Clin Biol Res       Date:  1977

8.  Characterization of unusual hexosaminidase A (HEX A) deficient human mutants.

Authors:  J S O'Brien; L Tennant; M L Veath; C R Scott; W E Bucknall
Journal:  Am J Hum Genet       Date:  1978-11       Impact factor: 11.025

9.  Nonuniform deficiency of hexosaminidase A in tissues and fluids of two unrelated individuals.

Authors:  G H Thomas; S Raghavan; E H Kolodny; A Frisch; E F Neufeld; J S O'Brien; L W Reynolds; C S Miller; J Shapiro; H H Kazazian; R H Heller
Journal:  Pediatr Res       Date:  1982-03       Impact factor: 3.756

10.  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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  3 in total

Review 1.  "Pseudodeficiencies" of lysosomal hydrolases.

Authors:  G H Thomas
Journal:  Am J Hum Genet       Date:  1994-06       Impact factor: 11.025

2.  A pseudodeficiency allele common in non-Jewish Tay-Sachs carriers: implications for carrier screening.

Authors:  B L Triggs-Raine; E H Mules; M M Kaback; J S Lim-Steele; C E Dowling; B R Akerman; M R Natowicz; E E Grebner; R Navon; J P Welch
Journal:  Am J Hum Genet       Date:  1992-10       Impact factor: 11.025

3.  A second mutation associated with apparent beta-hexosaminidase A pseudodeficiency: identification and frequency estimation.

Authors:  Z Cao; M R Natowicz; M M Kaback; J S Lim-Steele; E M Prence; D Brown; T Chabot; B L Triggs-Raine
Journal:  Am J Hum Genet       Date:  1993-12       Impact factor: 11.025

  3 in total

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