Phosphatidic acid mediates demyelination in Lpin1 mutant mice

Genes Dev. 2008 Jun 15;22(12):1647-61. doi: 10.1101/gad.1638008.

Abstract

Lipids play crucial roles in many aspects of glial cell biology, affecting processes ranging from myelin membrane biosynthesis to axo-glial interactions. In order to study the role of lipid metabolism in myelinating glial cells, we specifically deleted in Schwann cells the Lpin1 gene, which encodes the Mg2+-dependent phosphatidate phosphatase (PAP1) enzyme necessary for normal triacylglycerol biosynthesis. The affected animals developed pronounced peripheral neuropathy characterized by myelin degradation, Schwann cell dedifferentiation and proliferation, and a reduction in nerve conduction velocity. The observed demyelination is mediated by endoneurial accumulation of the substrate of the PAP1 enzyme, phosphatidic acid (PA). In addition, we show that PA is a potent activator of the MEK-Erk pathway in Schwann cells, and that this activation is required for PA-induced demyelination. Our results therefore reveal a surprising role for PA in Schwann cell fate determination and provide evidence of a direct link between diseases affecting lipid metabolism and abnormal Schwann cell function.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Animals, Newborn
  • Cell Differentiation / genetics
  • Cells, Cultured
  • Demyelinating Diseases / etiology*
  • Demyelinating Diseases / genetics
  • Demyelinating Diseases / metabolism
  • Gene Expression Regulation
  • Mice
  • Mice, Inbred BALB C
  • Mice, Knockout
  • Myelin Sheath / metabolism
  • Nuclear Proteins / genetics*
  • Organ Specificity / genetics
  • Pancreatitis-Associated Proteins
  • Peripheral Nerves / metabolism
  • Phosphatidate Phosphatase
  • Phosphatidic Acids / physiology*
  • Protein Isoforms / genetics
  • Protein Isoforms / physiology
  • Proteins / genetics
  • Proteins / metabolism
  • Rats
  • Rats, Sprague-Dawley
  • Schwann Cells / metabolism
  • Schwann Cells / physiology

Substances

  • Nuclear Proteins
  • Pancreatitis-Associated Proteins
  • Phosphatidic Acids
  • Protein Isoforms
  • Proteins
  • REG3A protein, human
  • Reg3b protein, mouse
  • Lpin1 protein, mouse
  • Phosphatidate Phosphatase