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Reversal of neurobehavioral social deficits in dystrophic mice using inhibitors of phosphodiesterases PDE5A and PDE9A

Duchenne muscular dystrophy is caused by mutations in the DYSTROPHIN gene. Although primarily associated with muscle wasting, a significant portion of patients (approximately 25%) are also diagnosed with autism spectrum disorder. We describe social behavioral deficits in dystrophin-deficient mice an...

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Detalles Bibliográficos
Autores principales: Alexander, M S, Gasperini, M J, Tsai, P T, Gibbs, D E, Spinazzola, J M, Marshall, J L, Feyder, M J, Pletcher, M T, Chekler, E L P, Morris, C A, Sahin, M, Harms, J F, Schmidt, C J, Kleiman, R J, Kunkel, L M
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group 2016
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5048211/
https://www.ncbi.nlm.nih.gov/pubmed/27676442
http://dx.doi.org/10.1038/tp.2016.174
Descripción
Sumario:Duchenne muscular dystrophy is caused by mutations in the DYSTROPHIN gene. Although primarily associated with muscle wasting, a significant portion of patients (approximately 25%) are also diagnosed with autism spectrum disorder. We describe social behavioral deficits in dystrophin-deficient mice and present evidence of cerebellar deficits in cGMP production. We demonstrate therapeutic potential for selective inhibitors of the cGMP-specific PDE5A and PDE9A enzymes to restore social behaviors in dystrophin-deficient mice.