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An engineered AAV targeting integrin alpha V beta 6 presents improved myotropism across species.
Vu Hong, Ai; Suel, Laurence; Petat, Eva; Dubois, Auriane; Le Brun, Pierre-Romain; Guerchet, Nicolas; Veron, Philippe; Poupiot, Jérôme; Richard, Isabelle.
Afiliación
  • Vu Hong A; Genethon, 1 bis rue de l'internationale, Evry, France. avuhong@genethon.fr.
  • Suel L; INTEGRARE research unit UMR_S951 (INSERM, Université Paris-Saclay, Univ Evry), Evry, France. avuhong@genethon.fr.
  • Petat E; Genethon, 1 bis rue de l'internationale, Evry, France.
  • Dubois A; INTEGRARE research unit UMR_S951 (INSERM, Université Paris-Saclay, Univ Evry), Evry, France.
  • Le Brun PR; Genethon, 1 bis rue de l'internationale, Evry, France.
  • Guerchet N; INTEGRARE research unit UMR_S951 (INSERM, Université Paris-Saclay, Univ Evry), Evry, France.
  • Veron P; Genethon, 1 bis rue de l'internationale, Evry, France.
  • Poupiot J; INTEGRARE research unit UMR_S951 (INSERM, Université Paris-Saclay, Univ Evry), Evry, France.
  • Richard I; Genethon, 1 bis rue de l'internationale, Evry, France.
Nat Commun ; 15(1): 7965, 2024 Sep 11.
Article en En | MEDLINE | ID: mdl-39261465
ABSTRACT
Current adeno-associated virus (AAV) gene therapy using nature-derived AAVs is limited by non-optimal tissue targeting. In the treatment of muscular diseases (MD), high doses are often required but can lead to severe adverse effects. Here, we rationally design an AAV capsid that specifically targets skeletal muscle to lower treatment doses. We computationally integrate binding motifs of human integrin alphaV beta6, a skeletal muscle receptor, into a liver-detargeting capsid. Designed AAVs show higher productivity and superior muscle transduction compared to their parent. One variant, LICA1, demonstrates comparable muscle transduction to other myotropic AAVs with reduced liver targeting. LICA1's myotropic properties are observed across species, including non-human primate. Consequently, LICA1, but not AAV9, effectively delivers therapeutic transgenes and improved muscle functionality in two mouse MD models (male mice) at a low dose (5E12 vg/kg). These results underline the potential of our design method for AAV engineering and LICA1 variant for MD gene therapy.
Asunto(s)

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Terapia Genética / Dependovirus / Músculo Esquelético Límite: Animals / Humans / Male Idioma: En Revista: Nat Commun Asunto de la revista: BIOLOGIA / CIENCIA Año: 2024 Tipo del documento: Article País de afiliación: Francia Pais de publicación: Reino Unido

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Terapia Genética / Dependovirus / Músculo Esquelético Límite: Animals / Humans / Male Idioma: En Revista: Nat Commun Asunto de la revista: BIOLOGIA / CIENCIA Año: 2024 Tipo del documento: Article País de afiliación: Francia Pais de publicación: Reino Unido