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Combined immunodeficiency due to purine nucleoside phosphorylase deficiency: Outcome of three patients.
Torun, Basak; Bilgin, Ahmet; Orhan, Diclehan; Gocmen, Rahsan; Kilic, Sebnem Sara; Kuskonmaz, Baris; Cetinkaya, Duygu; Tezcan, Ilhan; Cagdas, Deniz.
Afiliação
  • Torun B; Hacettepe University Medical School, Ankara, Turkey. Electronic address: basak.torun@hacettepe.edu.tr.
  • Bilgin A; Department of Pediatrics, Hacettepe University Medical School, Ankara, Turkey.
  • Orhan D; Departments of Pediatric and Perinatal Pathology, Hacettepe University Medical School, Ankara, Turkey.
  • Gocmen R; Department of Radiology, Hacettepe University Medical School, Ankara, Turkey.
  • Kilic SS; Department of Pediatric Immunology, Uludag University, School of Medicine, Bursa, Turkey.
  • Kuskonmaz B; Department of Pediatric Hematology, Hacettepe University Medical School, Ankara, Turkey.
  • Cetinkaya D; Department of Pediatric Hematology, Hacettepe University Medical School, Ankara, Turkey.
  • Tezcan I; Department of Pediatric Immunology, Hacettepe University Medical School, Ankara, Turkey.
  • Cagdas D; Department of Pediatric Immunology, Hacettepe University Medical School, Ankara, Turkey.
Eur J Med Genet ; 65(3): 104428, 2022 Mar.
Article em En | MEDLINE | ID: mdl-35063692
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.
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Texto completo: 1 Coleções: 01-internacional Temas: Geral Base de dados: MEDLINE Assunto principal: Erros Inatos do Metabolismo da Purina-Pirimidina / Doenças da Imunodeficiência Primária / Síndromes de Imunodeficiência Tipo de estudo: Diagnostic_studies Limite: Humans Idioma: En Revista: Eur j med genet Assunto da revista: GENETICA MEDICA Ano de publicação: 2022 Tipo de documento: Article

Texto completo: 1 Coleções: 01-internacional Temas: Geral Base de dados: MEDLINE Assunto principal: Erros Inatos do Metabolismo da Purina-Pirimidina / Doenças da Imunodeficiência Primária / Síndromes de Imunodeficiência Tipo de estudo: Diagnostic_studies Limite: Humans Idioma: En Revista: Eur j med genet Assunto da revista: GENETICA MEDICA Ano de publicação: 2022 Tipo de documento: Article