Literature DB >> 8673045

Late diagnosis and correction of purine nucleoside phosphorylase deficiency with allogeneic bone marrow transplantation.

P A Carpenter1, J B Ziegler, M R Vowels.   

Abstract

A 5-year-old boy with spastic quadriplegia, T cell immunodeficiency, hypouricemia and immune cytopenias from age 8 months, was found to have purine nucleoside phosphorylase (PNP) deficiency, and developed chronic lung disease. Successful matched sibling BMT for PNP deficiency has not previously been reported. BMT using marrow from an HLA-identical sibling donor was performed after conditioning with busulfan (16 mg/kg), cyclophosphamide (200 mg/kg), melphalan (90 mg/m2) and anti-thymocyte globulin (36 mg/kg). T lymphocyte numbers, PNP activity and uric acid levels rapidly improved and he remains well 12 months after transplant.

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Year:  1996        PMID: 8673045

Source DB:  PubMed          Journal:  Bone Marrow Transplant        ISSN: 0268-3369            Impact factor:   5.483


  7 in total

Review 1.  Stem cell transplantation for immunodeficiency.

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2.  Novel Genetic Mutations in the First Swedish Patient with Purine Nucleoside Phosphorylase Deficiency and Clinical Outcome After Hematopoietic Stem Cell Transplantation with HLA-Matched Unrelated Donor.

Authors:  Nicholas Brodszki; Maria Svensson; André B P van Kuilenburg; Judith Meijer; Lida Zoetekouw; Lennart Truedsson; Jacek Toporski
Journal:  JIMD Rep       Date:  2015-05-13

3.  Inborn errors of purine metabolism: clinical update and therapies.

Authors:  Shanti Balasubramaniam; John A Duley; John Christodoulou
Journal:  J Inherit Metab Dis       Date:  2014-06-28       Impact factor: 4.982

4.  Assessment of the red cell proteome of young patients with unexplained hemolytic anemia by two-dimensional differential in-gel electrophoresis (DIGE).

Authors:  Katharina von Löhneysen; Thomas M Scott; Katrin Soldau; Xiuling Xu; Jeffrey S Friedman
Journal:  PLoS One       Date:  2012-04-03       Impact factor: 3.240

5.  Changes in serum uric acid levels after allogeneic hematologic stem cell transplantation: A retrospective cohort study.

Authors:  Sang Hyun Joo; Jin Kyun Park; Eunyoung Emily Lee; Yeong Wook Song; Sung-Soo Yoon
Journal:  Blood Res       Date:  2016-09-23

6.  Infusion of Sibling Marrow in a Patient with Purine Nucleoside Phosphorylase Deficiency Leads to Split Mixed Donor Chimerism and Normal Immunity.

Authors:  Laura Yeates; Mary A Slatter; Andrew R Gennery
Journal:  Front Pediatr       Date:  2017-06-19       Impact factor: 3.418

7.  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

  7 in total

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