ArticleClinical and diagnostic laboratory immunology2005
polyethylene glycol-conjugated adenosine deaminase (ADA) therapy provides temporary immune reconstitution to a child with delayed-onset ADA deficiency.
Article in Clinical and diagnostic laboratory immunology, 2005. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers.
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The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
14 citing papers in PubMed, 31 citations in OpenAlex.
- Trial
- Innovative Design of Targeted Nanoparticles: Polymer-Drug Conjugates for Enhanced Cancer Therapy.Pharmaceutics · 2023Review
- Review
- Metabolite and thymocyte development defects in ADA-SCID mice receiving enzyme replacement therapy.Scientific reports · 2021Article
- From Synthesis to Characterization of Site-Selective PEGylated Proteins.Frontiers in pharmacology · 2019Review
- Gene Therapy for Adenosine Deaminase Deficiency: A Comprehensive Evaluation of Short- and Medium-Term Safety.Molecular therapy : the journal of the American Society of Gene Therapy · 2018Article
- Diagnosis, Treatment and Long-Term Follow Up of Patients with ADA Deficiency: a Single-Center Experience.Journal of clinical immunology · 2015Article
- Adenosine-associated delivery systems.Journal of drug targeting · 2015Review
- Autoimmune dysregulation and purine metabolism in adenosine deaminase deficiency.Frontiers in immunology · 2012Article
- Recombinant adeno-associated virus-mediated gene transfer for the potential therapy of adenosine deaminase-deficient severe combined immune deficiency.Human gene therapy · 2011Article
- How I treat ADA deficiency.Blood · 2009Review
- Pegademase bovine (PEG-ADA) for the treatment of infants and children with severe combined immunodeficiency (SCID).Biologics : targets & therapy · 2009Article
- TAT-mediated intracellular delivery of purine nucleoside phosphorylase corrects its deficiency in mice.The Journal of clinical investigation · 2006Article
- Increasing importance of stem cell gene therapy in adenosine deaminase deficiency?Clinical and vaccine immunology : CVI · 2006Article
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Authors and funding
8 authors at 4 institutions in 2 countries.
Funding
Abstract
We describe the effects of polyethylene glycol-conjugated adenosine deaminase (ADA) replacement therapy on lymphocyte counts, activation, apoptosis, proliferation, and cytokine secretion in a 14-month-old girl with "delayed-onset" ADA deficiency and marked immunodysregulation. Pretreatment lymphopenia affected T cells (CD4, 150/microl; CD8, 459/microl), B cells (16/microl), and NK cells (55/microl). T cells were uniformly activated and largely apoptotic (CD4, 59%; CD8, 82%); and T-cell-dependent cytokine levels in plasma were elevated, including the levels of interleukin 2 (IL-2; 26 pg/ml), IL-4 (81 pg/ml), IL-5 (46 pg/ml), gamma interferon (1,430 pg/ml), tumor necrosis factor alpha (210 pg/ml), and IL-10 (168 pg/ml). Mitogen-stimulated peripheral blood mononuclear cells show reduced IL-2 secretion and proliferation. During the first 5 months of therapy there was clinical improvement and partial immune reconstitution, with nearly normal lymphocyte subset numbers, reduced T-cell activation and CD4-cell apoptosis, and decreased plasma cytokine levels. In parallel, IL-2 secretion and the lymphocyte mitogenic response improved. Between 4 and 7 months, immunoglobulin G antibodies to bovine ADA developed and resulted in the complete reversal of immune recovery.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.