| Literature DB >> 15509508 |
Loïc Dupré1, Sara Trifari, Antonia Follenzi, Francesco Marangoni, Teresa Lain de Lera, Antonio Bernad, Silvana Martino, Shigeru Tsuchiya, Claudio Bordignon, Luigi Naldini, Alessandro Aiuti, Maria-Grazia Roncarolo.
Abstract
Wiskott-Aldrich syndrome (WAS) is an X-linked primary immunodeficiency with a median survival below the age of 20 due to infections, severe hemorrhage, and lymphomas. Transplantation of hematopoietic stem cells from HLA-identical sibling donors is a resolutive treatment, but is available for a minority of patients. Transplantation of genetically corrected autologous hematopoietic stem cells or T cells could represent an alternative treatment applicable to all patients. We investigated whether WAS gene transfer with MMLV-based oncoretroviral and HIV-based lentiviral vectors could restore normal functions of patients' T cells. T cells transduced either with lentiviral vectors expressing the WAS protein (WASP) from the ubiquitous PGK promoter or the tissue-specific WASP promoter or with an oncoretroviral vector expressing WASP from the LTR, reached normal levels of WASP with correction of functional defects, including proliferation, IL-2 production, and lipid raft upregulation. Lentiviral vectors transduced T cells from WAS patients at higher rates, compared to oncoretroviral vectors, and efficiently transduced both activated and naive WAS T cells. Furthermore, a selective growth advantage of T cells corrected with the lentiviral vectors was demonstrated. The observation that lentiviral vector-mediated gene transfer results in correction of T cell defects in vitro supports their application for gene therapy in WAS patients.Entities:
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Year: 2004 PMID: 15509508 DOI: 10.1016/j.ymthe.2004.08.008
Source DB: PubMed Journal: Mol Ther ISSN: 1525-0016 Impact factor: 11.454