The transcription factor Nurr1 is upregulated in amyotrophic lateral sclerosis patients and SOD1-G93A mice

Dis Model Mech. 2020 May 15;13(5):dmm043513. doi: 10.1242/dmm.043513.

Abstract

Amyotrophic lateral sclerosis (ALS) is a neurodegenerative disease that affects both lower and upper motor neurons (MNs) in the central nervous system. ALS etiology is highly multifactorial and multifarious, and an effective treatment is still lacking. Neuroinflammation is a hallmark of ALS and could be targeted to develop new therapeutic approaches. Interestingly, the transcription factor Nurr1 has been demonstrated to have an important role in the inflammatory process in several neurological disorders, such as Parkinson's disease and multiple sclerosis. In the present paper, we demonstrate for the first time that Nurr1 expression levels are upregulated in the peripheral blood of ALS patients. Moreover, we investigated Nurr1 function in the SOD1-G93A mouse model of ALS. Nurr1 was strongly upregulated in the spinal cord during the asymptomatic and early symptomatic phases of the disease, where it promoted the expression of brain-derived neurotrophic factor mRNA and the repression of NFκB pro-inflammatory targets, such as inducible nitric oxide synthase. Therefore, we hypothesize that Nurr1 is activated in an early phase of the disease as a protective endogenous anti-inflammatory mechanism, although not sufficient to reverse disease progression. On the basis of these observations, Nurr1 could represent a potential biomarker for ALS and a promising target for future therapies.

Keywords: ALS; Motor neuron disease; Neuroinflammation; Nurr1; SOD1-G93A mice.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Amyotrophic Lateral Sclerosis / blood
  • Amyotrophic Lateral Sclerosis / genetics*
  • Animals
  • Astrocytes / metabolism
  • Astrocytes / pathology
  • Brain-Derived Neurotrophic Factor / metabolism
  • Female
  • Gene Expression Regulation
  • Humans
  • Male
  • Mice
  • Mice, Transgenic
  • Middle Aged
  • Motor Neurons / metabolism
  • Motor Neurons / pathology
  • NF-kappa B / genetics
  • NF-kappa B / metabolism
  • Nitric Oxide Synthase Type II / genetics
  • Nitric Oxide Synthase Type II / metabolism
  • Nuclear Receptor Subfamily 4, Group A, Member 2 / blood
  • Nuclear Receptor Subfamily 4, Group A, Member 2 / genetics*
  • Nuclear Receptor Subfamily 4, Group A, Member 2 / metabolism
  • Promoter Regions, Genetic / genetics
  • RNA, Messenger / genetics
  • RNA, Messenger / metabolism
  • Spinal Cord / metabolism
  • Spinal Cord / pathology
  • Superoxide Dismutase-1 / genetics*
  • Transcription Factors / genetics*
  • Transcription Factors / metabolism
  • Transcriptional Activation / genetics
  • Up-Regulation / genetics*

Substances

  • Brain-Derived Neurotrophic Factor
  • NF-kappa B
  • Nuclear Receptor Subfamily 4, Group A, Member 2
  • RNA, Messenger
  • Transcription Factors
  • Nitric Oxide Synthase Type II
  • Superoxide Dismutase-1