| Literature DB >> 31880951 |
Laura Muraine1, Mona Bensalah1, Jamila Dhiab1, Gonzalo Cordova1, Ludovic Arandel1, Alix Marhic1, Maud Chapart2, Stéphane Vasseur2, Sofia Benkhelifa-Ziyyat1, Anne Bigot1, Gillian Butler-Browne1, Vincent Mouly1, Elisa Negroni1, Capucine Trollet1.
Abstract
The adeno-associated virus (AAV) vector is an efficient tool for gene delivery in skeletal muscle. AAV-based therapies show promising results for treatment of various genetic disorders, including muscular dystrophy. These dystrophies represent a heterogeneous group of diseases affecting muscles and typically characterized by progressive skeletal muscle wasting and weakness and the development of fibrosis. The tropism of each AAV serotype has been extensively studied using systemic delivery routes, but very few studies have compared their transduction efficiency through direct intramuscular injection. Yet, in some muscular dystrophies, where only a few muscles are primarily affected, a local intramuscular injection to target these muscles would be the most appropriate route. A comprehensive comparison between different recombinant AAV (rAAV) serotypes is therefore needed. In this study, we investigated the transduction efficiency of rAAV serotypes 1-10 by local injection in skeletal muscle of control C57BL/6 mice. We used a CMV-nls-LacZ reporter cassette allowing nuclear expression of LacZ to easily localize targeted cells. Detection of β-galactosidase activity on muscle cryosections demonstrated that rAAV serotypes 1, 7, 8, 9, and 10 were more efficient than the others, with rAAV9 being the most efficient in mice. Furthermore, using a model of human muscle xenograft in immunodeficient mice, we observed that in human muscle, rAAV8 and rAAV9 had similar transduction efficiency. These findings demonstrate for the first time that the human muscle xenograft can be used to evaluate AAV-based therapeutical approaches in a human context.Entities:
Keywords: AAV; fibrosis; preclinical; skeletal muscle; tropism; xenograft
Mesh:
Year: 2020 PMID: 31880951 PMCID: PMC7047108 DOI: 10.1089/hum.2019.173
Source DB: PubMed Journal: Hum Gene Ther ISSN: 1043-0342 Impact factor: 5.695