A Translational Pathway Toward a Clinical Trial Using the Second-Generation AAV Micro-Dystrophin Vector

Abstract

Duchenne muscular dystrophy (DMD) is a life threatening disease affecting all muscles in the body. An important therapeutic goal of DMD gene therapy is to deliver a therapeutic gene to all muscles in the body. The overarching goal of this project is to achieve systemic AAV-8 mediated expression of a low immunogenic human micro-dystrophin gene to young adult affected dogs. In this funding period, we demonstrated that we can achieve efficient whole body skeletal muscle and heart gene transfer in neonatal dogs with AAV-8 by a single intravenous injection. We also showed that systemic delivery of a caninemicro-dystrophin AAV vector is safe in young adult affected dogs. These results established a solid foundation to test systemic low-immunogenic human microgene delivery in young adult affected dogs in subsequent years of this project. In addition, we have performed a comprehensive review on the current status of DMD gene therapy in the canine model. We also contributed another article and reviewed the use of animal models in the development of DMD gene therapy.

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Document Details

Document Type
Technical Report
Publication Date
Sep 01, 2015
Accession Number
AD1007822

Entities

People

  • Chady H. Hakim
  • Craig A Emter
  • Dongsheng Duan
  • Hsiao T. Yang
  • Yi Lai
  • Yongping Yue

Organizations

  • University of Missouri System

Tags

DTIC Thesaurus Topics

  • Biochemistry
  • Biological Staining And Labeling
  • Biomedical Research
  • Body Weight
  • Cells
  • Clinical Trials
  • Demographic Cohorts
  • Department Of Defense
  • Engineering
  • Gene Delivery
  • Gene Therapy
  • Genes
  • Genetics
  • Muscles
  • Skeletal Muscle
  • Therapy
  • Tissues

Fields of Study

  • Biology
  • Medicine

Readers

  • Immunology and Pathology
  • Molecular Genetics

Technology Areas

  • Biotechnology