Literature DB >> 111549

Cellular immune deficiency with autoimmune hemolytic anemia in purine nucleoside phosphorylase deficiency.

K C Rich, W J Arnold, T Palella, I H Fox.   

Abstract

Immunologic and metabolic abnormalities were studied in a five year old boy with 0.07 per cent of normal erythrocyte purine nucleoside phosphorylase activity. The clinical course is characterized by severe autoimmune hemolytic anemia, a transient neurologic disorder with tremor and ataxia, and minor infectious illnesses. There is severe lymphopenia with decreased absolute numbers of T and B lymphocytes. Mitogen-stimulated blastogenesis is reduced, but response to allogeneic lymphocytes is normal. A monoclonal IgG protein is present. There is hypouricemia, elevated plasma inosine level, hypouricosuria and an increase in the urinary concentration of inosine and guanosine. The pattern of heterozygote distribution in the patient's family is compatible with an autosomal recessive trait in which heterozygotes are identifiable. In addition, the unusual laboratory and clinical manifestations of this patient illustrate the heterogeneity of the clinical syndrome associated with purine nucleoside phosphorylase deficiency.

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Year:  1979        PMID: 111549     DOI: 10.1016/0002-9343(79)90100-1

Source DB:  PubMed          Journal:  Am J Med        ISSN: 0002-9343            Impact factor:   4.965


  9 in total

1.  Point mutations at the purine nucleoside phosphorylase locus impair thymocyte differentiation in the mouse.

Authors:  F F Snyder; J P Jenuth; E R Mably; R K Mangat
Journal:  Proc Natl Acad Sci U S A       Date:  1997-03-18       Impact factor: 11.205

Review 2.  Structural analyses reveal two distinct families of nucleoside phosphorylases.

Authors:  Matthew J Pugmire; Steven E Ealick
Journal:  Biochem J       Date:  2002-01-01       Impact factor: 3.857

3.  Purine enzymes in patients with rheumatoid arthritis treated with methotrexate.

Authors:  A E van Ede; R F J M Laan; R A De Abreu; A B J Stegeman; L B A van de Putte
Journal:  Ann Rheum Dis       Date:  2002-12       Impact factor: 19.103

4.  Purinogenic immunodeficiency diseases. Differential effects of deoxyadenosine and deoxyguanosine on DNA synthesis in human T lymphoblasts.

Authors:  J M Wilson; B S Mitchell; P E Daddona; W N Kelley
Journal:  J Clin Invest       Date:  1979-11       Impact factor: 14.808

5.  Altered purine and pyrimidine metabolism in erythrocytes with purine nucleoside phosphorylase deficiency.

Authors:  I H Fox; J Kaminska; N L Edwards; E Gelfand; K C Rich; W N Arnold
Journal:  Biochem Genet       Date:  1980-04       Impact factor: 1.890

6.  B cell hyperactivity and abnormalities in T cell markers and immunoregulatory function in a patient with nucleoside phosphorylase deficiency.

Authors:  J M Zabay; E G De La Concha; C Ludeña; C Lozano; D Pascual-Salcedo; A Bootello; P Gonzalezporqué
Journal:  Clin Exp Immunol       Date:  1982-12       Impact factor: 4.330

7.  Deficiency of purine nucleoside phosphorylase activity in thymocytes from the immunodeficient diabetic BB rat.

Authors:  G Wu; E B Marliss
Journal:  Clin Exp Immunol       Date:  1991-11       Impact factor: 4.330

Review 8.  Lymphopenia, Lymphopenia-Induced Proliferation, and Autoimmunity.

Authors:  Ting-Ting Sheu; Bor-Luen Chiang
Journal:  Int J Mol Sci       Date:  2021-04-16       Impact factor: 5.923

9.  Partial Purine Nucleoside Phosphorylase Deficiency Helps Determine Minimal Activity Required for Immune and Neurological Development.

Authors:  Eyal Grunebaum; Nicholas Campbell; Matilde Leon-Ponte; Xiaobai Xu; Hugo Chapdelaine
Journal:  Front Immunol       Date:  2020-06-30       Impact factor: 7.561

  9 in total

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