Literature DB >> 6863546

Genetic heterogeneity in partial adenosine deaminase deficiency.

R Hirschhorn, F Martiniuk, V Roegner-Maniscalco, A Ellenbogen, J L Perignon, T Jenkins.   

Abstract

Inherited deficiency of the enzyme adenosine deaminase (ADA) results in a syndrome of severe combined immunodeficiency (SCID). Children with ADA- -SCID lack ADA in all cells and tissues. In contrast, a "partial" deficiency of ADA has been described in six immunologically normal children from four different "families." These children lack ADA in their erythrocytes but retain variable amounts of activity in their lymphoid cells. We have examined ADA activity in lymphoid line cells from four of these children, who are unrelated, for evidence of genetic heterogeneity. One child, who is Caucasian, has an enzyme with increased electrophoretic mobility, a diminished isoelectric point (pI 4.8 vs. Nl = 4.9) and very low activity (2.3 vs. Nl = 82.9 +/- 12.9 nmol/mg protein per min); as a second child has an enzyme with normal electrophoretic mobility but increased isoelectric point (pI = 5.0), markedly diminished heat stability at 56 degrees C (t1/2 = 4.2' vs. Nl = 40') and low activity (12.1); a third has an enzyme with only diminished heat stability (t1/2 = 6.5'), no detectable abnormality in charge and almost normal activity (41.9); while the fourth exhibits only diminished ADA activity (25.0) with no striking qualitative abnormalities. Thus, we have found evidence for three different mutations at the structural locus for ADA in three of these individuals, (a) an acidic, low activity heat stable mutation (b) a basic, somewhat higher activity, heat labile mutation, and (c) a relatively normal activity heat labile mutation. In the fourth, there is as yet no compelling evidence for a mutation at the structural locus for ADA and a mutation at a regulatory locus cannot be excluded.

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Year:  1983        PMID: 6863546      PMCID: PMC370394          DOI: 10.1172/jci110944

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  24 in total

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Authors:  M S Coleman; J J Hutton
Journal:  Biochem Med       Date:  1975-05

2.  Assignment of a gene for adenosine deaminase to human chromosome 20.

Authors:  J A Tischfield; R P Creagan; E A Nichols; F H Ruddle
Journal:  Hum Hered       Date:  1974       Impact factor: 0.444

3.  Red-blood-cell adenosine deaminase deficiency in a "healthy" Kung individual.

Authors:  T Jenkins
Journal:  Lancet       Date:  1973-09-29       Impact factor: 79.321

4.  Adenosine-deaminase deficiency in two patients with severely impaired cellular immunity.

Authors:  E R Giblett; J E Anderson; F Cohen; B Pollara; H J Meuwissen
Journal:  Lancet       Date:  1972-11-18       Impact factor: 79.321

5.  Evidence for control of several different tissue-specific isozymes of adenosine deaminase by a single genetic locus.

Authors:  R Hirschhorn; V Levytaka; B Pollara; H J Meuwissen
Journal:  Nat New Biol       Date:  1973-12-19

6.  The investigation of reactive sulphydryls in enzymes and their variants by starch gel electrophoresis. Studies on red cell adenosine deaminase.

Authors:  D A Hopkinson; H Harris
Journal:  Ann Hum Genet       Date:  1969-07       Impact factor: 1.670

7.  Conversion of human erythrocyte-adenosine deaminase activity to different tissue-specific isozymes. Evidence for a common catalytic unit.

Authors:  R Hirschhorn
Journal:  J Clin Invest       Date:  1975-03       Impact factor: 14.808

8.  Deficiency of adenosine deaminase not associated with severe combined immunodeficiency.

Authors:  T Jenkins; A R Rabson; G T Nurse; A B Lane
Journal:  J Pediatr       Date:  1976-11       Impact factor: 4.406

9.  Enzyme replacement therapy for adenosine deaminase deficiency and severe combined immunodeficiency.

Authors:  S H Polmar; R C Stern; A L Schwartz; E M Wetzler; P A Chase; R Hirschhorn
Journal:  N Engl J Med       Date:  1976-12-09       Impact factor: 91.245

10.  Further data on the adenosine deaminase (ADA) polymprphism and a report of a new phenotype.

Authors:  D A Hopkinson; P J Cook; H Harris
Journal:  Ann Hum Genet       Date:  1969-05       Impact factor: 1.670

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  12 in total

1.  Identification of a point mutation resulting in a heat-labile adenosine deaminase (ADA) in two unrelated children with partial ADA deficiency.

Authors:  R Hirschhorn; S Tzall; A Ellenbogen; S H Orkin
Journal:  J Clin Invest       Date:  1989-02       Impact factor: 14.808

2.  Structure of adenosine deaminase mRNAs from normal and adenosine deaminase-deficient human cell lines.

Authors:  G S Adrian; D A Wiginton; J J Hutton
Journal:  Mol Cell Biol       Date:  1984-09       Impact factor: 4.272

3.  Sequence of human adenosine deaminase cDNA including the coding region and a small intron.

Authors:  D A Wiginton; G S Adrian; J J Hutton
Journal:  Nucleic Acids Res       Date:  1984-03-12       Impact factor: 16.971

4.  Hot spot mutations in adenosine deaminase deficiency.

Authors:  R Hirschhorn; S Tzall; A Ellenbogen
Journal:  Proc Natl Acad Sci U S A       Date:  1990-08       Impact factor: 11.205

5.  Partial adenosine deaminase deficiency: another family from southern Africa.

Authors:  S L Hart; A B Lane; T Jenkins
Journal:  Hum Genet       Date:  1986-11       Impact factor: 4.132

6.  Transient expression of human adenosine deaminase cDNAs: identification of a nonfunctional clone resulting from a single amino acid substitution.

Authors:  S H Orkin; S C Goff; W N Kelley; P E Daddona
Journal:  Mol Cell Biol       Date:  1985-04       Impact factor: 4.272

7.  Identification of a point mutation in the adenosine deaminase gene responsible for immunodeficiency.

Authors:  D T Bonthron; A F Markham; D Ginsburg; S H Orkin
Journal:  J Clin Invest       Date:  1985-08       Impact factor: 14.808

8.  Genetic heterogeneity in adenosine deaminase (ADA) deficiency: five different mutations in five new patients with partial ADA deficiency.

Authors:  R Hirschhorn; A Ellenbogen
Journal:  Am J Hum Genet       Date:  1986-01       Impact factor: 11.025

9.  Characterization of normal and mutant adenosine deaminase messenger RNAs by translation and hybridization to a cDNA probe.

Authors:  G S Adrian; D A Wiginton; J J Hutton
Journal:  Hum Genet       Date:  1984       Impact factor: 4.132

10.  Paradoxical expression of adenosine deaminase in T cells cultured from a patient with adenosine deaminase deficiency and combine immunodeficiency.

Authors:  F X Arredondo-Vega; J Kurtzberg; S Chaffee; I Santisteban; E Reisner; M S Povey; M S Hershfield
Journal:  J Clin Invest       Date:  1990-08       Impact factor: 14.808

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