Olfactory deficit and gastrointestinal dysfunction precede motor abnormalities in alpha-Synuclein G51D knock-in mice

Proc Natl Acad Sci U S A. 2024 Sep 24;121(39):e2406479121. doi: 10.1073/pnas.2406479121. Epub 2024 Sep 16.

Abstract

Parkinson's disease (PD) is typically a sporadic late-onset disorder, which has made it difficult to model in mice. Several transgenic mouse models bearing mutations in SNCA, which encodes alpha-Synuclein (α-Syn), have been made, but these lines do not express SNCA in a physiologically accurate spatiotemporal pattern, which limits the ability of the mice to recapitulate the features of human PD. Here, we generated knock-in mice bearing the G51D SNCA mutation. After establishing that their motor symptoms begin at 9 mo of age, we then sought earlier pathologies. We assessed the phosphorylation at Serine 129 of α-Syn in different tissues and detected phospho-α-Syn in the olfactory bulb and enteric nervous system at 3 mo of age. Olfactory deficit and impaired gut transit followed at 6 mo, preceding motor symptoms. The SncaG51D mice thus parallel the progression of human PD and will enable us to study PD pathogenesis and test future therapies.

Keywords: G51D mutants; Parkinson’s disease; alpha-Synuclein; gastrointestinal dysfunction; olfactory deficit.

MeSH terms

  • Animals
  • Disease Models, Animal*
  • Enteric Nervous System / metabolism
  • Enteric Nervous System / physiopathology
  • Gastrointestinal Diseases / genetics
  • Gastrointestinal Diseases / metabolism
  • Gastrointestinal Diseases / pathology
  • Gene Knock-In Techniques*
  • Humans
  • Male
  • Mice
  • Mice, Transgenic
  • Olfaction Disorders / genetics
  • Olfaction Disorders / metabolism
  • Olfaction Disorders / physiopathology
  • Olfactory Bulb / metabolism
  • Olfactory Bulb / pathology
  • Parkinson Disease* / genetics
  • Parkinson Disease* / metabolism
  • Parkinson Disease* / pathology
  • Parkinson Disease* / physiopathology
  • Phosphorylation
  • alpha-Synuclein* / genetics
  • alpha-Synuclein* / metabolism

Substances

  • alpha-Synuclein
  • Snca protein, mouse