Dixdc1 is a critical regulator of DISC1 and embryonic cortical development

Neuron. 2010 Jul 15;67(1):33-48. doi: 10.1016/j.neuron.2010.06.002.

Abstract

The psychiatric illness risk gene Disrupted in Schizophrenia-1 (DISC1) plays an important role in brain development; however, it is unclear how DISC1 is regulated during cortical development. Here, we report that DISC1 is regulated during embryonic neural progenitor proliferation and neuronal migration through an interaction with DIX domain containing-1 (Dixdc1), the third mammalian gene discovered to contain a Disheveled-Axin (DIX) domain. We determined that Dixdc1 functionally interacts with DISC1 to regulate neural progenitor proliferation by co-modulating Wnt-GSK3beta/beta-catenin signaling. However, DISC1 and Dixdc1 do not regulate migration via this pathway. During neuronal migration, we discovered that phosphorylation of Dixdc1 by cyclin-dependent kinase 5 (Cdk5) facilitates its interaction with the DISC1-binding partner Ndel1. Furthermore, Dixdc1 phosphorylation and its interaction with DISC1/Ndel1 in vivo is required for neuronal migration. Together, these data reveal that Dixdc1 integrates DISC1 into Wnt-GSK3beta/beta-catenin-dependent and -independent signaling pathways during cortical development and further delineate how DISC1 contributes to neuropsychiatric disorders.

Publication types

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

MeSH terms

  • Animals
  • Animals, Newborn
  • Bromodeoxyuridine / metabolism
  • Cell Cycle Proteins / genetics
  • Cell Cycle Proteins / metabolism
  • Cell Movement / genetics
  • Cell Movement / physiology
  • Cell Proliferation
  • Cells, Cultured
  • Cerebral Cortex / cytology
  • Cerebral Cortex / embryology*
  • Electroporation / methods
  • Embryo, Mammalian
  • Female
  • Gene Expression Regulation, Developmental / physiology
  • Green Fluorescent Proteins / genetics
  • Humans
  • Intracellular Signaling Peptides and Proteins / genetics
  • Intracellular Signaling Peptides and Proteins / metabolism*
  • Luminescent Proteins / genetics
  • Mice
  • Microfilament Proteins / genetics
  • Microfilament Proteins / metabolism*
  • Microtubule-Associated Proteins
  • Nerve Tissue Proteins / genetics
  • Nerve Tissue Proteins / metabolism*
  • Neurons / physiology*
  • Pregnancy
  • Protein Binding / genetics
  • RNA Interference / physiology
  • Signal Transduction / drug effects
  • Signal Transduction / genetics
  • Stem Cells / physiology
  • Transfection / methods
  • Wnt Proteins / genetics
  • Wnt Proteins / metabolism

Substances

  • Cell Cycle Proteins
  • DISC1 protein, human
  • DIXDC1 protein, human
  • Intracellular Signaling Peptides and Proteins
  • Luminescent Proteins
  • Microfilament Proteins
  • Microtubule-Associated Proteins
  • Nde1 protein, mouse
  • Nerve Tissue Proteins
  • Wnt Proteins
  • enhanced green fluorescent protein
  • Green Fluorescent Proteins
  • Bromodeoxyuridine