Literature DB >> 35063692

Combined immunodeficiency due to purine nucleoside phosphorylase deficiency: Outcome of three patients.

Basak Torun1, Ahmet Bilgin2, Diclehan Orhan3, Rahsan Gocmen4, Sebnem Sara Kılıc5, Barıs Kuskonmaz6, Duygu Cetinkaya6, Ilhan Tezcan7, Deniz Cagdas7.   

Abstract

Purine nucleoside phosphorylase (PNP) is a key enzyme in the purine salvage pathway. PNP deficiency, caused by the autosomal recessive mutations in the PNP gene, can lead to severe combined immunodeficiency (SCID). PNP deficiency patients typically have profound T-cell deficiency with variable B and NK cell functions. They present clinically with recurrent infections, failure to thrive, various neurological disorders, malignancies, and autoimmune diseases. Hematopoietic stem cell transplantation (HSCT) is the only available cure for patients with PNP deficiency. We present three patients, two of whom were successfully treated with HSCT. One patient died prior to HSCT due to EBV-associated lymphoma. Over the course of post-HSCT, there was no further aggravation of the patients' neurological symptoms. Although both of the patients still had mild developmental delay, new developmental milestones were achieved.
Copyright © 2022 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  Hematopoietic stem cell transplantation; Low uric acid; Purine nucleoside phosphorylase; Severe combined immunodeficiency; T cell deficiency

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Year:  2022        PMID: 35063692     DOI: 10.1016/j.ejmg.2022.104428

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  2 in total

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