PUBLICATION

The microcephaly gene aspm is involved in brain development in zebrafish

Authors
Kim, H.T., Lee, M.S., Choi, J.H., Jung, J.Y., Ahn, D.G., Yeo, S.Y., Choi, D.K., and Kim, C.H.
ID
ZDB-PUB-110609-13
Date
2011
Source
Biochemical and Biophysical Research Communications   409(4): 640-4 (Journal)
Registered Authors
Ahn, Dae-gwon, Choi, Jung-Hwa, Kim, Cheol-Hee, Kim, Hyun-Taek, Lee, Mi-Sun, Yeo, Sang-Yeob
Keywords
Microcephaly; ASPM; CNS; Mitotic arrest; Zebrafish
MeSH Terms
  • Amino Acid Sequence
  • Animals
  • Brain/embryology*
  • Cell Cycle
  • Cell Cycle Proteins/genetics*
  • Gene Knockdown Techniques
  • Humans
  • Microcephaly/genetics*
  • Mitosis/genetics
  • Molecular Sequence Data
  • Organ Size/genetics
  • Zebrafish/embryology*
  • Zebrafish/genetics
  • Zebrafish Proteins/genetics*
PubMed
21620798 Full text @ Biochem. Biophys. Res. Commun.
Abstract
MCPH is a neurodevelopmental disorder characterized by a global reduction in cerebral cortical volume. Homozygous mutation of the MCPH5 gene, also known as ASPM, is the most common cause of the MCPH phenotype. To elucidate the roles of ASPM during embryonic development, the zebrafish aspm was identified, which is specifically expressed in proliferating cells in the CNS. Morpholino-mediated knock-down of aspm resulted in a significant reduction in head size. Furthermore, aspm-deficient embryos exhibited a mitotic arrest during early development. These findings suggest that the reduction in brain size in MCPH might be caused by lack of aspm function in the mitotic cell cycle and demonstrate that the zebrafish can provide a model system for congenital diseases of the human nervous system.
Genes / Markers
Figures
Expression
Phenotype
Mutation and Transgenics
Human Disease / Model Data
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping
Errata and Notes