AAV dose-dependent transduction efficiency in retinal ganglion cells and functional efficacy of optogenetic vision restoration

Gene Ther. 2024 Nov;31(11-12):572-579. doi: 10.1038/s41434-024-00485-7. Epub 2024 Sep 5.

Abstract

Optogenetics is a promising approach for restoring vision to the blind after photoreceptor degeneration. The ability to restore vision through AAV-mediated delivery of light-sensitive proteins, especially channelrhodopsins, into retinal ganglion cells has been extensively demonstrated in animal models. For clinical application, knowledge of viral dose-dependent functional efficacy is desired. In this study, using a triple-knockout blind mouse model and a highly light-sensitive channelrhodopsin variant, we evaluated viral dose-dependent vision restoration through retinal ganglion cell expression by using optomotor behavioral assays. Our results show that both the restored light sensitivity and visual acuity reached peak levels at a medial viral dose of 108 vg. With increasing dose, transduction efficiency continued to increase while protein expression peaked at the dose of ~109 vg and declined at higher doses. Also, a significant increase in retinal gliosis and inflammatory responses started at the dose of ~109 vg, and a marked increase was observed at the dose of ~1010. These results provide valuable insights into viral dose design for clinical studies.

MeSH terms

  • Animals
  • Blindness / genetics
  • Blindness / therapy
  • Channelrhodopsins / genetics
  • Channelrhodopsins / metabolism
  • Dependovirus* / genetics
  • Disease Models, Animal
  • Genetic Therapy* / methods
  • Genetic Vectors* / administration & dosage
  • Genetic Vectors* / genetics
  • Mice
  • Mice, Knockout*
  • Optogenetics* / methods
  • Retinal Degeneration / genetics
  • Retinal Degeneration / metabolism
  • Retinal Degeneration / therapy
  • Retinal Ganglion Cells* / metabolism
  • Transduction, Genetic* / methods
  • Vision, Ocular
  • Visual Acuity

Substances

  • Channelrhodopsins