Plerixafor+G-CSF-mobilized CD34+ cells represent an optimal graft source for thalassemia gene therapy

Blood. 2015 Jul 30;126(5):616-9. doi: 10.1182/blood-2015-03-629618. Epub 2015 Jun 18.

Abstract

Globin gene therapy requires abundant numbers of highly engraftable, autologous hematopoietic stem cells expressing curative levels of β-globin on differentiation. In this study, CD34+ cells from 31 thalassemic patients mobilized with hydroxyurea+granulocyte colony-stimulating factor (G-CSF), G-CSF, Plerixafor, or Plerixafor+G-CSF were transduced with the TNS9.3.55 β-globin lentivector and compared for transducibility and globin expression in vitro, as well as engraftment potential in a xenogeneic model after partial myeloablation. Transduction efficiency and vector copy number (VCN) averaged 48.4 ± 2.8% and 1.91 ± 0.04, respectively, whereas expression approximated the one-copy normal β-globin output. Plerixafor+G-CSF cells produced the highest β-globin expression/VCN. Long-term multilineage engraftment and persistent VCN and vector expression was encountered in all xenografted groups, with Plerixafor+G-CSF-mobilized cells achieving superior short-term engraftment rates, with similar numbers of CD34+ cells transplanted. Overall, Plerixafor+G-CSF not only allows high CD34+ cell yields but also provides increased β-globin expression/VCN and enhanced early human chimerism under nonmyeloablative conditions, thus representing an optimal graft for thalassemia gene therapy.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Antigens, CD34 / metabolism
  • Benzylamines
  • Cyclams
  • Gene Dosage
  • Gene Expression
  • Genetic Therapy / methods*
  • Genetic Vectors
  • Granulocyte Colony-Stimulating Factor / administration & dosage*
  • Hematopoietic Stem Cell Mobilization / methods*
  • Hematopoietic Stem Cell Transplantation*
  • Hematopoietic Stem Cells / metabolism
  • Heterocyclic Compounds / administration & dosage
  • Heterografts
  • Humans
  • Mice
  • Mice, Knockout
  • Transplantation, Autologous
  • beta-Globins / genetics
  • beta-Thalassemia / genetics
  • beta-Thalassemia / therapy*

Substances

  • Antigens, CD34
  • Benzylamines
  • Cyclams
  • Heterocyclic Compounds
  • beta-Globins
  • Granulocyte Colony-Stimulating Factor
  • plerixafor