AAV5 Delivery of CRISPR/Cas9 Mediates Genome Editing in the Lungs of Young Rhesus Monkeys

Hum Gene Ther. 2024 Oct;35(19-20):814-824. doi: 10.1089/hum.2024.035. Epub 2024 Jul 3.

Abstract

Genome editing has the potential to treat genetic diseases in a variety of tissues, including the lung. We have previously developed and validated a dual adeno-associated virus (AAV) CRISPR platform that supports effective editing in the airways of mice. To validate this delivery vehicle in a large animal model, we have shown that intratracheal instillation of CRISPR/Cas9 in AAV5 can edit a housekeeping gene or a disease-related gene in the lungs of young rhesus monkeys. We observed up to 8% editing of angiotensin-converting enzyme 2 (ACE2) in lung lobes after single-dose administration. Single-nuclear RNA sequencing revealed that AAV5 transduces multiple cell types in the caudal lung lobes, including alveolar cells, macrophages, fibroblasts, endothelial cells, and B cells. These results demonstrate that AAV5 is efficient in the delivery of CRISPR/Cas9 in the lung lobes of young rhesus monkeys.

Keywords: AAV5; CRISPR; lung; rhesus monkey.

Publication types

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

MeSH terms

  • Angiotensin-Converting Enzyme 2 / genetics
  • Angiotensin-Converting Enzyme 2 / metabolism
  • Animals
  • CRISPR-Cas Systems*
  • Dependovirus* / genetics
  • Gene Editing* / methods
  • Genetic Therapy / methods
  • Genetic Vectors* / administration & dosage
  • Genetic Vectors* / genetics
  • Humans
  • Lung* / metabolism
  • Macaca mulatta*

Substances

  • Angiotensin-Converting Enzyme 2