Drosophila Lis1 is required for neuroblast proliferation, dendritic elaboration and axonal transport

Nat Cell Biol. 2000 Nov;2(11):776-83. doi: 10.1038/35041011.


Haplo-insufficiency of human Lis1 causes lissencephaly. Reduced Lis1 activity in both humans and mice results in a neuronal migration defect. Here we show that Drosophila Lis1 is highly expressed in the nervous system. Lis1 is essential for neuroblast proliferation and axonal transport, as shown by a mosaic analysis using a Lis1 null mutation. Moreover, it is cell-autonomously required for dendritic growth, branching and maturation. Analogous mosaic analysis shows that neurons containing a mutated cytoplasmic-dynein heavy chain (Dhc64C) exhibit phenotypes similar to Lis1 mutants. These results implicate Lis1 as a regulator of the microtubule cytoskeleton and show that it is important for diverse physiological functions in the nervous system.

Publication types

  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Animals
  • Animals, Genetically Modified
  • Axonal Transport / physiology*
  • Cell Differentiation
  • Cell Division
  • Central Nervous System / embryology
  • Central Nervous System / metabolism
  • Dendrites / physiology*
  • Drosophila
  • Drosophila Proteins
  • Microtubule-Associated Proteins / genetics
  • Microtubule-Associated Proteins / metabolism*
  • Microtubule-Associated Proteins / physiology
  • Mutagenesis
  • Neurons / physiology*


  • Drosophila Proteins
  • Lis-1 protein, Drosophila
  • Microtubule-Associated Proteins