Extensive Transduction and Enhanced Spread of a Modified AAV2 Capsid in the Non-human Primate CNS

Mol Ther. 2018 Oct 3;26(10):2418-2430. doi: 10.1016/j.ymthe.2018.07.008. Epub 2018 Jul 12.

Abstract

The present study was designed to characterize transduction of non-human primate brain and spinal cord with a modified adeno-associated virus serotype 2, incapable of binding to the heparan sulfate proteoglycan receptor, referred to as AAV2-HBKO. AAV2-HBKO was infused into the thalamus, intracerebroventricularly or via a combination of both intracerebroventricular and thalamic delivery. Thalamic injection of this modified vector encoding GFP resulted in widespread CNS transduction that included neurons in deep cortical layers, deep cerebellar nuclei, several subcortical regions, and motor neuron transduction in the spinal cord indicative of robust bidirectional axonal transport. Intracerebroventricular delivery similarly resulted in widespread cortical transduction, with one striking distinction that oligodendrocytes within superficial layers of the cortex were the primary cell type transduced. Robust motor neuron transduction was also observed in all levels of the spinal cord. The combination of thalamic and intracerebroventricular delivery resulted in transduction of oligodendrocytes in superficial cortical layers and neurons in deeper cortical layers. Several subcortical regions were also transduced. Our data demonstrate that AAV2-HBKO is a powerful vector for the potential treatment of a wide number of neurological disorders, and highlight that delivery route can significantly impact cellular tropism and pattern of CNS transduction.

Keywords: AAV vectors; CNS disorders; brain; capsid modification; gene therapy; nervous system; non-human primate; spinal cord; transduction; viral vector.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Axonal Transport / drug effects
  • Brain / drug effects
  • Brain / pathology
  • Capsid Proteins / administration & dosage
  • Capsid Proteins / genetics
  • Central Nervous System / drug effects
  • Central Nervous System / pathology
  • Dependovirus
  • Disease Models, Animal
  • Genetic Therapy*
  • Genetic Vectors / administration & dosage
  • Genetic Vectors / adverse effects*
  • Genetic Vectors / genetics
  • Heparan Sulfate Proteoglycans / administration & dosage
  • Heparan Sulfate Proteoglycans / genetics
  • Humans
  • Infusions, Intraventricular
  • Motor Neurons / drug effects
  • Neurons / drug effects*
  • Neurons / pathology
  • Parvovirinae / genetics*
  • Primates
  • Spinal Cord / drug effects*
  • Spinal Cord / pathology
  • Thalamus / drug effects

Substances

  • Capsid Proteins
  • Heparan Sulfate Proteoglycans

Supplementary concepts

  • Adeno-associated virus-2