Tailoring the AAV2 capsid vector for bone-targeting

Pediatr Res. 2018 Oct;84(4):545-551. doi: 10.1038/s41390-018-0095-8. Epub 2018 Oct 15.

Abstract

Background: Targeting specific tissues remains a major challenge to the promise of gene therapy. For example, several strategies have failed to target adeno-associated virus 2 (AAV2) vectors, to bone. We have evaluated in vitro and in vivo the affinity of an AAV2 vector to bone matrix, hydroxyapatite (HA) to treat Mucopolysacccharidosis IVA.

Methods: To increase vector affinity to HA, an aspartic acid octapeptide (D8) was inserted immediately after the N-terminal region of the VP2 capsid protein. The modified vector had physical titers and transduction efficiencies comparable to the unmodified vector.

Results: The bone-targeting vector had significantly higher HA affinity and vector genome copies in bone than the unmodified vector. The modified vector was also released from HA, and its enzyme activity in bone, 3 months post infusion, was 4.7-fold higher than the unmodified vector.

Conclusion: Inserting a bone-targeting peptide into the vector capsid increases gene delivery and expression in the bone without decreasing enzyme expression. This approach could be a novel strategy to treat systemic bone diseases.

Publication types

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

MeSH terms

  • Animals
  • Aspartic Acid / chemistry
  • Bone Marrow / metabolism
  • Bone and Bones / metabolism*
  • Brain / metabolism
  • Capsid
  • Capsid Proteins / chemistry*
  • Dependovirus
  • Durapatite / chemistry*
  • Gene Expression Profiling
  • Gene Transfer Techniques
  • Genetic Therapy
  • Genetic Vectors*
  • HEK293 Cells
  • Humans
  • Hydroxyapatites / chemistry
  • Liver / metabolism
  • Mice
  • Mice, Transgenic
  • Mucopolysaccharidosis IV / therapy*
  • Parvovirinae
  • Protein Domains
  • Transgenes

Substances

  • Capsid Proteins
  • Hydroxyapatites
  • Aspartic Acid
  • Durapatite

Supplementary concepts

  • Adeno-associated virus-2