Development of Orally Bioavailable Therapeutics by the Chloroplast Expression System to Counter Muscle Degeneration, Weakness, and Fibrosis in DMD

Abstract

Patients with DMD suffer from progressive muscle weakness and damage, resulting in fibrotic replacement. The goal of this project is to evaluate the therapeutic potential of the anti-fibrotic agents, ACE2/Ang(1-7), when produced in plants using a chloroplast expression system. Lyophilized plant material was delivered by oral gavage to the mdx mouse model for DMD. Initial studies were done to ensure that the plant material and protein was orally bioavailable. Further, additional studies confirmed that ACE2 protein accumulated in the circulation over the course of the treatment. Functional assessment of mice treated for 2 weeks showed improved strength in the diaphragm and EDL muscles. However, by 2 months of treatment the benefits were reduced back to untreated controls. This lack of long-term benefit was not due to compound delivery, which accumulated in the circulation over the course of the study. Instead, we believe that the muscle tissue compensated by down regulating the receptors that are sensitive to the heightened Ang(1-7)levels. Even though this therapeutic agent did not provide benefit, we assert that this delivery strategy may provide a new way to introduce therapeutic proteins for treating neuromuscular disease.

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Document Details

Document Type
Technical Report
Publication Date
Feb 01, 2018
Accession Number
AD1059201

Entities

People

  • Elisabeth Barton

Organizations

  • University of Florida

Tags

DTIC Thesaurus Topics

  • Age Groups
  • Algorithms
  • Anatomy
  • Biological Sciences
  • Biomedical Research
  • Body Weight
  • Closed Loop Systems
  • Department Of Defense
  • Diseases And Disorders
  • Drug Therapy
  • Fibrosis
  • Maryland
  • Materials
  • Muscles
  • Muscular Diseases
  • Musculoskeletal Physiology
  • Neuromuscular Diseases
  • Plastids
  • Production
  • Skeletal Muscle
  • Therapy
  • Tissues
  • Tobacco Plants
  • Universities

Fields of Study

  • Medicine

Readers

  • Immunology and Pathology
  • Toxicology/Environmental Toxicology