Defects in T-cell-mediated immunity to influenza virus in murine Wiskott-Aldrich syndrome are corrected by oncoretroviral vector-mediated gene transfer into repopulating hematopoietic cells

Blood. 2003 Nov 1;102(9):3108-16. doi: 10.1182/blood-2002-11-3489. Epub 2003 Jul 10.

Abstract

The Wiskott-Aldrich syndrome (WAS) is an X-linked disorder characterized by immune dysfunction, thrombocytopenia, and eczema. We used a murine model created by knockout of the WAS protein gene (WASP) to evaluate the potential of gene therapy for WAS. Lethally irradiated, male WASP- animals that received transplants of mixtures of wild type (WT) and WASP- bone marrow cells demonstrated enrichment of WT cells in the lymphoid and myeloid lineages with a progressive increase in the proportion of WT T-lymphoid and B-lymphoid cells. WASP- mice had a defective secondary T-cell response to influenza virus which was normalized in animals that received transplants of 35% or more WT cells. The WASP gene was inserted into WASP- bone marrow cells with a bicistronic oncoretroviral vector also encoding green fluorescent protein (GFP), followed by transplantation into irradiated male WASP- recipients. There was a selective advantage for gene-corrected cells in multiple lineages. Animals with higher proportions of GFP+ T cells showed normalization of their lymphocyte counts. Gene-corrected, blood T cells exhibited full and partial correction, respectively, of their defective proliferative and cytokine secretory responses to in vitro T-cell-receptor stimulation. The defective secondary T-cell response to influenza virus was also improved in gene-corrected animals.

Publication types

  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Animals
  • Bone Marrow Cells / cytology
  • Bone Marrow Cells / metabolism
  • Bone Marrow Transplantation
  • Genetic Therapy / methods*
  • Genetic Vectors
  • Hematopoietic Stem Cells / cytology
  • Hematopoietic Stem Cells / metabolism
  • Immunity, Cellular
  • Male
  • Mice
  • Mice, Knockout
  • Orthomyxoviridae / immunology*
  • Orthomyxoviridae Infections / complications
  • Orthomyxoviridae Infections / immunology
  • Proteins / administration & dosage*
  • Proteins / genetics
  • Retroviridae / genetics
  • T-Lymphocytes / immunology*
  • Transduction, Genetic / methods
  • Treatment Outcome
  • Wiskott-Aldrich Syndrome / complications
  • Wiskott-Aldrich Syndrome / immunology
  • Wiskott-Aldrich Syndrome / therapy*
  • Wiskott-Aldrich Syndrome Protein

Substances

  • Proteins
  • Was protein, mouse
  • Wiskott-Aldrich Syndrome Protein