Genetic ablation of P65 subunit of NF-κB in mdx mice to improve muscle physiological function

Muscle Nerve. 2017 Oct;56(4):759-767. doi: 10.1002/mus.25517. Epub 2017 May 15.

Abstract

Introduction: Duchenne muscular dystrophy (DMD) is a genetic muscle disease characterized by dystrophin deficiency. Beyond gene replacement, the question of whether ablation of the p65 gene of nuclear factor-kappa B (NF-κB) in DMD can improve muscle physiology function is unknown. In this study, we investigated muscle physiological improvement in mdx mice (DMD model) with a genetic reduction of NF-κB.

Methods: Muscle physiological function and histology were studied in 2-month-old mdx/p65+/- , wild-type, mdx, and human minidystrophin gene transgenic mdx (TghΔDys/mdx) mice.

Results: Improved muscle physiological function was found in mdx/p65+/- mice when compared with mdx mice; however, it was similar to TghΔDys/mdx mice. The results indicate that genetic reduction of p65 levels diminished chronic inflammation in dystrophic muscle, thus leading to amelioration of muscle pathology and improved muscle physiological function.

Conclusions: The results show that inhibition of NF-κB may be a promising therapy when combined with gene therapy for DMD. Muscle Nerve 56: 759-767, 2017.

Keywords: DMD; NF-κB pathway; NF-κB/p65 knockout mice; human dystrophin transgenic mice; muscle physiological function.

MeSH terms

  • Animals
  • Gene Deletion*
  • Humans
  • Mice
  • Mice, Inbred mdx
  • Mice, Transgenic
  • Muscle, Skeletal / pathology
  • Muscle, Skeletal / physiology*
  • Muscular Dystrophy, Duchenne / genetics*
  • Muscular Dystrophy, Duchenne / pathology
  • Muscular Dystrophy, Duchenne / physiopathology
  • NF-kappa B / deficiency
  • NF-kappa B / genetics*
  • Protein Subunits / deficiency
  • Protein Subunits / genetics*

Substances

  • NF-kappa B
  • Protein Subunits