Mixed chimeric hematopoietic stem cell transplant reverses the disease phenotype in canine leukocyte adhesion deficiency

Vet Immunol Immunopathol. 2003 Oct 15;95(3-4):113-21. doi: 10.1016/s0165-2427(03)00108-9.

Abstract

The genetic disease canine leukocyte adhesion deficiency (CLAD) is characterized by recurrent, severe bacterial infections, typically culminating in death by 6 months of age. CLAD is due to a mutation in the leukocyte integrin CD18 subunit, which prevents surface expression of the CD11/CD18 leukocyte integrin complex. We demonstrate that stable mixed donor:host hematopoietic chimerism, achieved by a non-myeloablative bone marrow transplant from a histocompatible littermate, reverses the disease phenotype in CLAD. Donor chimerism following the transplant was demonstrated both by flow cytometric detection of donor-derived CD18-positive leukocytes in the peripheral blood of the recipient, and by the demonstration of donor-derived DNA microsatellite repeats in the peripheral blood leukocytes of the recipient. These results indicate that mixed hematopoietic chimerism reverses the clinical phenotype in CLAD and represents a potential therapeutic approach for the human disease leukocyte adhesion deficiency.

MeSH terms

  • Animals
  • Antigens, CD34 / immunology
  • CD11 Antigens / immunology
  • CD18 Antigens / immunology
  • DNA / chemistry
  • DNA / genetics
  • Dog Diseases / immunology
  • Dog Diseases / pathology
  • Dog Diseases / therapy*
  • Dogs
  • Flow Cytometry / veterinary
  • Hematopoietic Stem Cell Transplantation / methods
  • Hematopoietic Stem Cell Transplantation / veterinary*
  • Leukocyte Count / veterinary
  • Leukocyte-Adhesion Deficiency Syndrome / immunology
  • Leukocyte-Adhesion Deficiency Syndrome / pathology
  • Leukocyte-Adhesion Deficiency Syndrome / therapy
  • Leukocyte-Adhesion Deficiency Syndrome / veterinary*
  • Polymerase Chain Reaction / veterinary
  • Transplantation Chimera / immunology*

Substances

  • Antigens, CD34
  • CD11 Antigens
  • CD18 Antigens
  • DNA