Literature DB >> 4815083

Detection of the carrier state in combined immunodeficiency disease associated with adenosine deaminase deficiency.

C R Scott, S H Chen, E R Giblett.   

Abstract

A large pedigree containing a child with severe combined immunodeficiency disease (CID) associated with adenosine deaminase (ADA) deficiency was investigated to ascertain if heterozygotes could be detected by measuring red cell ADA activity. 9 of 17 individuals in three generations who were at risk for being heterozygous had decreased red cell ADA activity. This genetic information establishes one form of CID as an autosomal recessive disorder. The identified heterozygote population had a mean ADA value of 19.2 U/g hemoglobin (0.95 confidence interval; 14.0 to 24.4 U/g hemoglobin), which was approximately one-half the mean, 36.1 U/g hemoglobin, of a randomly selected control population (0.95 confidence interval; 22.5-58.1 U/g hemoglobin). Statistical comparisons of the heterozygotes to the normal population indicates that within a high-risk family heterozygotes may be identified with 90% confidence.

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Year:  1974        PMID: 4815083      PMCID: PMC333106          DOI: 10.1172/JCI107658

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


  11 in total

1.  A newly defined X-linked trait in man with demonstration of the Lyon effect in carrier females.

Authors:  D B Windhorst; B Holmes; R A Good
Journal:  Lancet       Date:  1967-04-08       Impact factor: 79.321

2.  Letter: Severe combined immunodeficiency and adenosine-deaminase deficiency.

Authors:  R Hirschhorn; N G Beratis
Journal:  Lancet       Date:  1973-11-24       Impact factor: 79.321

3.  Multiple forms of human adenosine deaminase. II. Isolation and properties of a conversion factor from human lung.

Authors:  H Nishihara; S Ishikawa; K Shinkai; H Akedo
Journal:  Biochim Biophys Acta       Date:  1973-04-12

4.  Adenosine-deaminase deficiency and severe combined immunodeficiency syndrome.

Authors:  H D Ochs; J E Yount; E R Giblett; S H Chen; C R Scott; R J Wedgwood
Journal:  Lancet       Date:  1973-06-16       Impact factor: 79.321

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

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

6.  Adenosine deaminases of two different molecular sizes in human tissues.

Authors:  H Akedo; H Nishihara; K Shinkae; K Komatsu
Journal:  Biochim Biophys Acta       Date:  1970-07-15

7.  Adenosine deaminase isozymes in human tissues.

Authors:  Y H Edwards; D A Hopkinson; H Harris
Journal:  Ann Hum Genet       Date:  1971-10       Impact factor: 1.670

8.  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

9.  The pattern of genetic transmission of the leukocyte defect in fatal granulomatous disease of childhood.

Authors:  D B Windhorst; A R Page; B Holmes; P G Quie; R A Good
Journal:  J Clin Invest       Date:  1968-05       Impact factor: 14.808

10.  Pyrimidine starvation induced by adenosine in fibroblasts and lymphoid cells: role of adenosine deaminase.

Authors:  H Green; T Chan
Journal:  Science       Date:  1973-11-23       Impact factor: 47.728

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  16 in total

1.  Evaluation of adenosine deaminase and other purine salvage pathway enzymes in horses with combined immunodeficiency.

Authors:  T C McGuire; B Pollara; J J Moore; M J Poppie
Journal:  Infect Immun       Date:  1976-03       Impact factor: 3.441

Review 2.  The biochemical basis of immunodeficiency disease.

Authors:  K O Raivio
Journal:  Eur J Pediatr       Date:  1980-10       Impact factor: 3.183

3.  Purine nucleoside phosphorylase deficiency. Measurement of variant protein in four families with enzyme-deficient members by an enzyme-linked immunosorbent assay.

Authors:  W R Osborne; C R Scott
Journal:  Am J Hum Genet       Date:  1980-11       Impact factor: 11.025

4.  Adenosine deaminase activity in pleural effusions: an aid to differential diagnosis.

Authors:  M A Piras; C Gakis; M Budroni; G Andreoni
Journal:  Br Med J       Date:  1978 Dec 23-30

5.  Heterogeneity for adenosine deaminase deficiency: Expression of the enzyme in cultured skin fibroblasts and amniotic fluid cells.

Authors:  S H Chen; C R Scott; D R Swedberg
Journal:  Am J Hum Genet       Date:  1975-01       Impact factor: 11.025

6.  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

7.  Adenosine deaminase deficiency: disappearance of adenine deoxynucleotides from a patient's erythrocytes after successful marrow transplantation.

Authors:  S H Chen; H D Ochs; C R Scott; E R Giblett; A J Tingle
Journal:  J Clin Invest       Date:  1978-12       Impact factor: 14.808

8.  Purine nucleoside phosphorylase deficiency. Evidence for molecular heterogeneity in two families with enzyme-deficient members.

Authors:  W R Osborne; S H Chen; E R Giblett; W D Biggar; A A Ammann; C R Scott
Journal:  J Clin Invest       Date:  1977-09       Impact factor: 14.808

9.  New assignment of the adenosine deaminase gene locus to chromosome 20q13 X 11 by study of a patient with interstitial deletion 20q.

Authors:  M B Petersen; L Tranebjaerg; N Tommerup; P Nygaard; H Edwards
Journal:  J Med Genet       Date:  1987-02       Impact factor: 6.318

10.  A normal level of adenosine deaminase activity in the red cell lysates of carriers and patients with severe combined immunodeficiency disease.

Authors:  P P Trotta; E M Smithwick; M E Balis
Journal:  Proc Natl Acad Sci U S A       Date:  1976-01       Impact factor: 11.205

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