Literature DB >> 21067383

Stem-cell gene therapy for the Wiskott-Aldrich syndrome.

Kaan Boztug1, Manfred Schmidt, Adrian Schwarzer, Pinaki P Banerjee, Inés Avedillo Díez, Ricardo A Dewey, Marie Böhm, Ali Nowrouzi, Claudia R Ball, Hanno Glimm, Sonja Naundorf, Klaus Kühlcke, Rainer Blasczyk, Irina Kondratenko, László Maródi, Jordan S Orange, Christof von Kalle, Christoph Klein.   

Abstract

The Wiskott-Aldrich syndrome (WAS) is an X-linked recessive primary immunodeficiency disorder associated with thrombocytopenia, eczema, and autoimmunity. We treated two patients who had this disorder with a transfusion of autologous, genetically modified hematopoietic stem cells (HSC). We found sustained expression of WAS protein expression in HSC, lymphoid and myeloid cells, and platelets after gene therapy. T and B cells, natural killer (NK) cells, and monocytes were functionally corrected. After treatment, the patients' clinical condition markedly improved, with resolution of hemorrhagic diathesis, eczema, autoimmunity, and predisposition to severe infection. Comprehensive insertion-site analysis showed vector integration that targeted multiple genes controlling growth and immunologic responses in a persistently polyclonal hematopoiesis. (Funded by Deutsche Forschungsgemeinschaft and others; German Clinical Trials Register number, DRKS00000330.).

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Year:  2010        PMID: 21067383      PMCID: PMC3064520          DOI: 10.1056/NEJMoa1003548

Source DB:  PubMed          Journal:  N Engl J Med        ISSN: 0028-4793            Impact factor:   91.245


  25 in total

1.  Gene therapy for Wiskott-Aldrich syndrome: rescue of T-cell signaling and amelioration of colitis upon transplantation of retrovirally transduced hematopoietic stem cells in mice.

Authors:  Christoph Klein; Deanna Nguyen; Ching-Hui Liu; Atsushi Mizoguchi; Atul K Bhan; Hiroaki Miki; Tadaomi Takenawa; Fred S Rosen; Fred W Alt; Richard C Mulligan; Scott B Snapper
Journal:  Blood       Date:  2002-11-14       Impact factor: 22.113

2.  Isolation of a novel gene mutated in Wiskott-Aldrich syndrome.

Authors:  J M Derry; H D Ochs; U Francke
Journal:  Cell       Date:  1994-08-26       Impact factor: 41.582

Review 3.  WASP: a key immunological multitasker.

Authors:  Adrian J Thrasher; Siobhan O Burns
Journal:  Nat Rev Immunol       Date:  2010-03       Impact factor: 53.106

4.  Gene therapy of human severe combined immunodeficiency (SCID)-X1 disease.

Authors:  M Cavazzana-Calvo; S Hacein-Bey; G de Saint Basile; F Gross; E Yvon; P Nusbaum; F Selz; C Hue; S Certain; J L Casanova; P Bousso; F L Deist; A Fischer
Journal:  Science       Date:  2000-04-28       Impact factor: 47.728

5.  Correction of X-linked chronic granulomatous disease by gene therapy, augmented by insertional activation of MDS1-EVI1, PRDM16 or SETBP1.

Authors:  Marion G Ott; Manfred Schmidt; Kerstin Schwarzwaelder; Stefan Stein; Ulrich Siler; Ulrike Koehl; Hanno Glimm; Klaus Kühlcke; Andrea Schilz; Hana Kunkel; Sonja Naundorf; Andrea Brinkmann; Annette Deichmann; Marlene Fischer; Claudia Ball; Ingo Pilz; Cynthia Dunbar; Yang Du; Nancy A Jenkins; Neal G Copeland; Ursula Lüthi; Moustapha Hassan; Adrian J Thrasher; Dieter Hoelzer; Christof von Kalle; Reinhard Seger; Manuel Grez
Journal:  Nat Med       Date:  2006-04-02       Impact factor: 53.440

6.  Wiskott-Aldrich syndrome protein is required for NK cell cytotoxicity and colocalizes with actin to NK cell-activating immunologic synapses.

Authors:  Jordan S Orange; Narayanaswamy Ramesh; Eileen Remold-O'Donnell; Yoji Sasahara; Louise Koopman; Michael Byrne; Francisco A Bonilla; Fred S Rosen; Raif S Geha; Jack L Strominger
Journal:  Proc Natl Acad Sci U S A       Date:  2002-08-12       Impact factor: 11.205

7.  LMO2-associated clonal T cell proliferation in two patients after gene therapy for SCID-X1.

Authors:  S Hacein-Bey-Abina; C Von Kalle; M Schmidt; M P McCormack; N Wulffraat; P Leboulch; A Lim; C S Osborne; R Pawliuk; E Morillon; R Sorensen; A Forster; P Fraser; J I Cohen; G de Saint Basile; I Alexander; U Wintergerst; T Frebourg; A Aurias; D Stoppa-Lyonnet; S Romana; I Radford-Weiss; F Gross; F Valensi; E Delabesse; E Macintyre; F Sigaux; J Soulier; L E Leiva; M Wissler; C Prinz; T H Rabbitts; F Le Deist; A Fischer; M Cavazzana-Calvo
Journal:  Science       Date:  2003-10-17       Impact factor: 47.728

8.  Clinical course of patients with WASP gene mutations.

Authors:  Kohsuke Imai; Tomohiro Morio; Yi Zhu; Yinzhu Jin; Sukeyuki Itoh; Michiko Kajiwara; Jun-Ichi Yata; Shuki Mizutani; Hans D Ochs; Shigeaki Nonoyama
Journal:  Blood       Date:  2003-09-11       Impact factor: 22.113

9.  Correction of ADA-SCID by stem cell gene therapy combined with nonmyeloablative conditioning.

Authors:  Alessandro Aiuti; Shimon Slavin; Memet Aker; Francesca Ficara; Sara Deola; Alessandra Mortellaro; Shoshana Morecki; Grazia Andolfi; Antonella Tabucchi; Filippo Carlucci; Enrico Marinello; Federica Cattaneo; Sergio Vai; Paolo Servida; Roberto Miniero; Maria Grazia Roncarolo; Claudio Bordignon
Journal:  Science       Date:  2002-06-28       Impact factor: 47.728

10.  Deficiency in the Wiskott-Aldrich protein induces premature proplatelet formation and platelet production in the bone marrow compartment.

Authors:  Siham Sabri; Adlen Foudi; Siham Boukour; Brigitte Franc; Sabine Charrier; Martine Jandrot-Perrus; Richard W Farndale; Abdelali Jalil; Mike P Blundell; Elisabeth M Cramer; Fawzia Louache; Najet Debili; Adrian J Thrasher; William Vainchenker
Journal:  Blood       Date:  2006-03-07       Impact factor: 22.113

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  198 in total

1.  Cutaneous manifestations of DOCK8 deficiency syndrome.

Authors:  Emily Y Chu; Alexandra F Freeman; Huie Jing; Edward W Cowen; Joie Davis; Helen C Su; Steven M Holland; Maria L Chanco Turner
Journal:  Arch Dermatol       Date:  2011-09-19

2.  Gene therapy for metabolic disorders: an overview with a focus on urea cycle disorders.

Authors:  Ian E Alexander; Cindy Kok; Allison P Dane; Sharon C Cunningham
Journal:  J Inherit Metab Dis       Date:  2012-03-09       Impact factor: 4.982

Review 3.  Hematopoietic stem cell engineering at a crossroads.

Authors:  Isabelle Rivière; Cynthia E Dunbar; Michel Sadelain
Journal:  Blood       Date:  2011-11-17       Impact factor: 22.113

4.  Lentiviral vector integration in the human genome induces alternative splicing and generates aberrant transcripts.

Authors:  Arianna Moiani; Ylenia Paleari; Daniela Sartori; Riccardo Mezzadra; Annarita Miccio; Claudia Cattoglio; Fabienne Cocchiarella; Maria Rosa Lidonnici; Giuliana Ferrari; Fulvio Mavilio
Journal:  J Clin Invest       Date:  2012-04-23       Impact factor: 14.808

5.  Gene therapy: too much splice can spoil the dish.

Authors:  Didier Trono
Journal:  J Clin Invest       Date:  2012-04-23       Impact factor: 14.808

6.  Gene therapies advance towards finish line.

Authors:  Asher Mullard
Journal:  Nat Rev Drug Discov       Date:  2011-09-30       Impact factor: 84.694

7.  Novel therapies, high-risk pediatric research, and the prospect of benefit: learning from the ethical disagreements.

Authors:  Inmaculada de Melo-Martín; Dolan Sondhi; Ronald G Crystal
Journal:  Mol Ther       Date:  2012-06       Impact factor: 11.454

8.  Advances in foamy virus vector technology and disease correction could speed the path to clinical application.

Authors:  Els Verhoeyen
Journal:  Mol Ther       Date:  2012-06       Impact factor: 11.454

Review 9.  Gene therapy for hemoglobinopathies: the state of the field and the future.

Authors:  Shanmuganathan Chandrakasan; Punam Malik
Journal:  Hematol Oncol Clin North Am       Date:  2014-04       Impact factor: 3.722

Review 10.  The transformative potential of HSC gene therapy as a genetic medicine.

Authors:  Pervinder Sagoo; H Bobby Gaspar
Journal:  Gene Ther       Date:  2021-05-26       Impact factor: 5.250

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