Leucine-Rich Repeat Kinase-2 Controls the Differentiation and Maturation of Oligodendrocytes in Mice and Zebrafish

Biomolecules. 2024 Jul 19;14(7):870. doi: 10.3390/biom14070870.

Abstract

Leucine-rich repeat kinase-2 (LRRK2), a gene mutated in familial and sporadic Parkinson's disease (PD), controls multiple cellular processes important for GLIA physiology. Interestingly, emerging studies report that LRRK2 is highly expressed in oligodendrocyte precursor cells (OPCs) compared to the pathophysiology of other brain cells and oligodendrocytes (OLs) in PD. Altogether, these observations suggest crucial function(s) of LRRK2 in OPCs/Ols, which would be interesting to explore. In this study, we investigated the role of LRRK2 in OLs. We showed that LRRK2 knock-out (KO) OPC cultures displayed defects in the transition of OPCs into OLs, suggesting a role of LRRK2 in OL differentiation. Consistently, we found an alteration of myelin basic protein (MBP) striosomes in LRRK2 KO mouse brains and reduced levels of oligodendrocyte transcription factor 2 (Olig2) and Mbp in olig2:EGFP and mbp:RFP transgenic zebrafish embryos injected with lrrk2 morpholino (MO). Moreover, lrrk2 knock-down zebrafish exhibited a lower amount of nerve growth factor (Ngf) compared to control embryos, which represents a potent regulator of oligodendrogenesis and myelination. Overall, our findings indicate that LRRK2 controls OL differentiation, affecting the number of mature OLs.

Keywords: LRRK2; Parkinson’s disease; oligodendrocytes; zebrafish.

MeSH terms

  • Animals
  • Animals, Genetically Modified
  • Cell Differentiation* / genetics
  • Leucine-Rich Repeat Serine-Threonine Protein Kinase-2* / genetics
  • Leucine-Rich Repeat Serine-Threonine Protein Kinase-2* / metabolism
  • Mice
  • Mice, Knockout
  • Myelin Basic Protein / genetics
  • Myelin Basic Protein / metabolism
  • Oligodendroglia* / cytology
  • Oligodendroglia* / metabolism
  • Zebrafish Proteins* / genetics
  • Zebrafish Proteins* / metabolism
  • Zebrafish* / genetics
  • Zebrafish* / metabolism

Substances

  • Leucine-Rich Repeat Serine-Threonine Protein Kinase-2
  • Lrrk2 protein, mouse
  • Zebrafish Proteins
  • Myelin Basic Protein
  • LRRK2 protein, zebrafish

Grants and funding

We are grateful for the financial support of Regional Foundation for Biomedical Research (FRRB, grant ID: 1737591, IR), MJF Foundation (MJFF-021665 to IR) and to the Italian Ministry of Health (Ricerca Corrente 2023, MG).