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Updated: May 6, 2026

Live Imaging of Mitosis in the Developing Mouse Embryonic Cortex
Published on: June 4, 2014
The microcephaly protein Asp regulates neuroepithelium morphogenesis by controlling the spatial distribution of
Maria A Rujano1, Luis Sanchez-Pulido, Carole Pennetier
1UMR144, CNRS- Institut Curie, 12 rue Lhomond, 75005 Paris, France.
Abstract:
Mutations in ASPM are the most frequent cause of microcephaly, a disorder characterized by reduced brain size at birth. ASPM is recognized as a major regulator of brain size, yet its role during neural development remains poorly understood. Moreover, the role of ASPM proteins in invertebrate brain morphogenesis has never been investigated. Here, we characterized the function of the Drosophila ASPM orthologue, Asp, and found that asp mutants present severe defects in brain size and neuroepithelium morphogenesis. We show that size reduction depends on the mitotic function of Asp, whereas regulation of tissue shape depends on an uncharacterized function. Asp interacts with myosin II regulating its polarized distribution along the apico-basal axis. In the absence of Asp, mislocalization of myosin II results in interkinetic nuclear migration and tissue architecture defects. We propose that Asp regulates neuroepithelium morphogenesis through myosin-II-mediated structural and mechanical processes to maintain force balance and tissue cohesiveness.
Insights
Mutations in ASPM cause microcephaly. Drosophila Asp regulates brain size and neuroepithelium shape by interacting with myosin II, impacting nuclear migration and tissue architecture.
Area of Science:
- Developmental Biology
- Neuroscience
- Genetics
Background:
- Mutations in ASPM are a primary cause of microcephaly, a condition of reduced brain size.
- ASPM's precise role in neural development and invertebrate brain morphogenesis is not well understood.
Purpose of the Study:
- To investigate the function of the Drosophila ASPM orthologue, Asp.
- To elucidate Asp's role in brain size regulation and neuroepithelium morphogenesis.
Main Methods:
- Characterization of Drosophila asp mutants.
- Analysis of neuroepithelium morphogenesis and mitotic function.
- Investigation of Asp's interaction with myosin II and its effect on polarized distribution.
Main Results:
- Asp mutants exhibit significant defects in brain size and neuroepithelium morphogenesis.
- Brain size reduction is linked to Asp's mitotic function.
- Asp regulates tissue shape via interaction with myosin II, affecting its apico-basal distribution.
- Loss of Asp leads to mislocalization of myosin II, causing defects in interkinetic nuclear migration and tissue architecture.
Conclusions:
- Asp is crucial for both brain size control and neuroepithelium morphogenesis in Drosophila.
- Asp regulates tissue shape and architecture through myosin II-dependent structural and mechanical processes.
- Asp maintains force balance and tissue cohesiveness during neurodevelopment.
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