Enhancing atrial-specific gene expression using a calsequestrin cis-regulatory module 4 with a sarcolipin promoter

J Gene Med. 2018 Dec;20(12):e3060. doi: 10.1002/jgm.3060. Epub 2018 Dec 4.

Abstract

Background: Cardiac gene therapy using the adeno-associated virus serotype 9 vector is widely used because of its efficient transduction. However, the promoters used to drive expression often cause off-target localization. To overcome this, studies have applied cardiac-specific promoters, although expression is debilitated compared to that of ubiquitous promoters. To address these issues in the context of atrial-specific gene expression, an enhancer calsequestrin cis-regulatory module 4 (CRM4) and the highly atrial-specific promoter sarcolipin were combined to enhance expression and minimize off tissue expression.

Methods: To observe expression and bio-distribution, constructs were generated using two different reporter genes: luciferase and enhanced green fluorescent protein (EGFP). The ubiquitous cytomegalovirus (CMV), sarcolipin (SLN) and CRM4 combined with sarcolipin (CRM4.SLN) were compared and analyzed using the luciferase assay, western blotting, a quantitative polymerase chain reaction and fluorescence imaging.

Results: The CMV promoter containing vectors showed the strongest expression in vitro and in vivo. However, the module SLN combination showed enhanced atrial expression and a minimized off-target effect even when compared with the individual SLN promoter.

Conclusions: For gene therapy involving atrial gene transfer, the CRM4.SLN combination is a promising alternative to the use of the CMV promoter. CRM4.SLN had significant atrial expression and minimized extra-atrial expression.

Keywords: AAV9; CRM4; atrium; cis-acting regulatory module; gene therapy; sarcolipin.

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

  • Animals
  • Calsequestrin / genetics*
  • Calsequestrin / metabolism
  • Cytomegalovirus / genetics
  • Dependovirus / genetics
  • Gene Expression Regulation*
  • Genetic Therapy / methods
  • Genetic Vectors / genetics
  • Green Fluorescent Proteins / genetics
  • Green Fluorescent Proteins / metabolism
  • Heart Atria / metabolism*
  • Heart Failure / genetics
  • Heart Failure / therapy
  • Humans
  • Luciferases / genetics
  • Luciferases / metabolism
  • Mice
  • Muscle Proteins / genetics*
  • Muscle Proteins / metabolism
  • Promoter Regions, Genetic / genetics*
  • Proteolipids / genetics*
  • Proteolipids / metabolism
  • Transfection

Substances

  • Calsequestrin
  • Muscle Proteins
  • Proteolipids
  • enhanced green fluorescent protein
  • sarcolipin
  • Green Fluorescent Proteins
  • Luciferases