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Cellular immune deficiency with autoimmune hemolytic anemia in purine nucleoside phosphorylase deficiency.
Authors:K C Rich  W J Arnold  T Palella  I H Fox
Institution:Chicago Illinois, USA;Ann Arbor, Michigan, USA
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.
Keywords:Requests for reprints should be addressed to Dr  Moo Nahm Yum  Department of Pathology  Indiana University Medical Center  1100 West Michigan Street  Indianapolis  Indiana 46202  
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