Purified Protein Delivery to Activate an Epigenetically Silenced Allele in Mouse Brain

Methods Mol Biol. 2018:1767:227-239. doi: 10.1007/978-1-4939-7774-1_12.

Abstract

The ability to activate or repress specific genes in the brain could have a tremendous impact for understanding and treating neurological disorders. Artificial transcription factors based on zinc finger, TALE, and CRISPR/Cas9 programmable DNA-binding platforms have been widely used to regulate the expression of specific genes in cultured cells, but their delivery into the brain represents a critical challenge to apply such tools in live animals. In previous work, we developed a purified, zinc finger-based artificial transcription factor that could be injected systemically, cross the blood-brain barrier, and alter expression of a specific gene in the brain of an adult mouse model of Angelman syndrome. Importantly, our mode of delivery produced widespread distribution throughout the brain. Here we describe our most current methods for the production and purification of the factor, dosage optimization, and use of live animal fluorescence imaging to visualize the kinetics of distribution.

Keywords: Animal models; Engineered zinc finger protein; Gene therapy; Neurologic disease; Preclinical studies.

Publication types

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

MeSH terms

  • Alleles*
  • Animals
  • Brain / metabolism*
  • Gene Silencing*
  • Injections
  • Mice
  • Mice, Inbred C57BL
  • Optical Imaging / methods
  • Transcription Factors / administration & dosage*
  • Transcription Factors / chemistry
  • Transcription Factors / pharmacokinetics
  • Transcriptional Activation*
  • Zinc Fingers

Substances

  • Transcription Factors