Effects of transient immunosuppression on adenoassociated, virus-mediated, liver-directed gene transfer in rhesus macaques and implications for human gene therapy

Blood. 2006 Nov 15;108(10):3321-8. doi: 10.1182/blood-2006-04-017913. Epub 2006 Jul 25.

Abstract

In a clinical study of recombinant adeno-associated virus-2 expressing human factor IX (AAV2-FIX), we detected 2 impediments to long-term gene transfer. First, preexisting anti-AAV neutralizing antibodies (NABs) prevent vector from reaching the target tissue, and second, CD8(+) T-cell responses to hepatocyte-cell surface displayed AAV-capsid-terminated FIX expression after several weeks. Because the vector is incapable of synthesizing viral proteins, a short course of immunosuppression, until AAV capsid is cleared from the transduced cells, may mitigate the host T-cell response, allowing long-term expression of FIX. To evaluate coad-ministration of immunosuppression, we studied AAV8 vector infusion in rhesus macaques, natural hosts for AAV8. We administered AAV8-FIX in 16 macaques via the hepatic artery and assessed the effects of (1) preexisting anti-AAV8 NABs, (2) a standard T-cell immunosuppressive regimen, and (3) efficacy and safety of AAV8-FIX. We found that low titers (1:5) of preexisting NABs abrogate transduction, whereas animals with undetectable NABs are safely and effectively transduced by AAV8-FIX. Coadministration of mycophenolate mofetil and tacrolimus with vector does not induce toxicity and does not impair AAV transduction or FIX synthesis. These findings enable a clinical study to assess the effects of immunomodulation on long-term FIX expression in patients with hemophilia B.

Publication types

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

MeSH terms

  • Animals
  • Antibodies / pharmacology
  • Dependovirus* / genetics
  • Dependovirus* / immunology
  • Drug Therapy, Combination
  • Factor IX / administration & dosage
  • Factor IX / immunology
  • Gene Transfer Techniques
  • Genetic Therapy / methods*
  • Genetic Vectors / immunology
  • Genetic Vectors / pharmacokinetics
  • Hemophilia B / therapy*
  • Humans
  • Immunosuppression Therapy / methods*
  • Immunosuppression Therapy / standards
  • Liver / metabolism*
  • Macaca mulatta
  • Male
  • Mice
  • Mycophenolic Acid / administration & dosage
  • Mycophenolic Acid / analogs & derivatives
  • Organ Specificity
  • Tacrolimus / administration & dosage

Substances

  • Antibodies
  • Factor IX
  • Mycophenolic Acid
  • Tacrolimus