Literature DB >> 15509508

Lentiviral vector-mediated gene transfer in T cells from Wiskott-Aldrich syndrome patients leads to functional correction.

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.

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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


  27 in total

Review 1.  Advances in the field of lentivector-based transduction of T and B lymphocytes for gene therapy.

Authors:  Cecilia Frecha; Camille Lévy; François-Loïc Cosset; Els Verhoeyen
Journal:  Mol Ther       Date:  2010-08-24       Impact factor: 11.454

Review 2.  Gene therapy of metabolic diseases.

Authors:  Alain Fischer; Salima Hacein-Bey-Abina; Marina Cavazzana-Calvo
Journal:  J Inherit Metab Dis       Date:  2006 Apr-Jun       Impact factor: 4.982

3.  Lentivector Knockdown of CCR5 in Hematopoietic Stem and Progenitor Cells Confers Functional and Persistent HIV-1 Resistance in Humanized Mice.

Authors:  Renier Myburgh; Sandra Ivic; Michael S Pepper; Gustavo Gers-Huber; Duo Li; Annette Audigé; Mary-Aude Rochat; Vincent Jaquet; Stephan Regenass; Markus G Manz; Patrick Salmon; Karl-Heinz Krause; Roberto F Speck
Journal:  J Virol       Date:  2015-04-22       Impact factor: 5.103

Review 4.  Engineering Stem Cells for Biomedical Applications.

Authors:  Perry T Yin; Edward Han; Ki-Bum Lee
Journal:  Adv Healthc Mater       Date:  2015-03-13       Impact factor: 9.933

5.  Ubiquitous high-level gene expression in hematopoietic lineages provides effective lentiviral gene therapy of murine Wiskott-Aldrich syndrome.

Authors:  Alexander Astrakhan; Blythe D Sather; Byoung Y Ryu; Socheath Khim; Swati Singh; Stephanie Humblet-Baron; Hans D Ochs; Carol H Miao; David J Rawlings
Journal:  Blood       Date:  2012-03-19       Impact factor: 22.113

Review 6.  Gene transfer into hematopoietic stem cells as treatment for primary immunodeficiency diseases.

Authors:  Fabio Candotti
Journal:  Int J Hematol       Date:  2014-02-01       Impact factor: 2.490

7.  Lentiviral hematopoietic stem cell gene therapy in patients with Wiskott-Aldrich syndrome.

Authors:  Alessandro Aiuti; Luca Biasco; Samantha Scaramuzza; Francesca Ferrua; Maria Pia Cicalese; Cristina Baricordi; Francesca Dionisio; Andrea Calabria; Stefania Giannelli; Maria Carmina Castiello; Marita Bosticardo; Costanza Evangelio; Andrea Assanelli; Miriam Casiraghi; Sara Di Nunzio; Luciano Callegaro; Claudia Benati; Paolo Rizzardi; Danilo Pellin; Clelia Di Serio; Manfred Schmidt; Christof Von Kalle; Jason Gardner; Nalini Mehta; Victor Neduva; David J Dow; Anne Galy; Roberto Miniero; Andrea Finocchi; Ayse Metin; Pinaki P Banerjee; Jordan S Orange; Stefania Galimberti; Maria Grazia Valsecchi; Alessandra Biffi; Eugenio Montini; Anna Villa; Fabio Ciceri; Maria Grazia Roncarolo; Luigi Naldini
Journal:  Science       Date:  2013-07-11       Impact factor: 47.728

8.  Evidence for long-term efficacy and safety of gene therapy for Wiskott-Aldrich syndrome in preclinical models.

Authors:  Francesco Marangoni; Marita Bosticardo; Sabine Charrier; Elena Draghici; Michela Locci; Samantha Scaramuzza; Cristina Panaroni; Maurilio Ponzoni; Francesca Sanvito; Claudio Doglioni; Marie Liabeuf; Bernard Gjata; Marie Montus; Katherine Siminovitch; Alessandro Aiuti; Luigi Naldini; Loïc Dupré; Maria Grazia Roncarolo; Anne Galy; Anna Villa
Journal:  Mol Ther       Date:  2009-03-03       Impact factor: 11.454

9.  Comparison of insulators and promoters for expression of the Wiskott-Aldrich syndrome protein using lentiviral vectors.

Authors:  Rachel M Koldej; Gael Carney; Matthew M Wielgosz; Sheng Zhou; Jun Zhan; Brian P Sorrentino; Arthur W Nienhuis
Journal:  Hum Gene Ther Clin Dev       Date:  2013-06-20       Impact factor: 5.032

Review 10.  Wiskott-Aldrich Syndrome: Immunodeficiency resulting from defective cell migration and impaired immunostimulatory activation.

Authors:  Gerben Bouma; Siobhan O Burns; Adrian J Thrasher
Journal:  Immunobiology       Date:  2009-07-22       Impact factor: 3.144

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