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Related Experiment Video

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Mosmo Is Required for Zebrafish Craniofacial Formation.

Carlos Camacho-Macorra1,2, Marcos Sintes1, Noemí Tabanera1,2

  • 1Centro de Biología Molecular Severo Ochoa, Consejo Superior de Investigaciones Científicas, Universidad Autónoma de Madrid, Madrid, Spain.

Frontiers in Cell and Developmental Biology
|November 8, 2021
PubMed
Summary

Mosmo (Modulator of Smoothened) protein regulates Hedgehog signaling by degrading Smoothened. In zebrafish, inactivating Mosmo paralogs causes craniofacial defects, suggesting a role in human congenital malformations.

Keywords:
MosmoSmoothened (Smo)craniofacial abnormalitieshedgehog signaling (Hh)tetraspan transmembrane protein

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Area of Science:

  • Developmental Biology
  • Molecular Biology
  • Genetics

Background:

  • Hedgehog (Hh) signaling is crucial for development and homeostasis.
  • Mutations in Hh pathway genes lead to congenital malformations.
  • Mosmo (Modulator of Smoothened) is a tetraspan protein that down-regulates Hh signaling by promoting Smoothened (Smo) degradation.

Purpose of the Study:

  • To investigate the role of Mosmo in vertebrate embryonic development.
  • To determine if Mosmo's modulation of Hh signaling is essential for embryonic development.

Main Methods:

  • Studied the expression of two zebrafish mosmo paralogs (mosmoa and mosmob) in embryonic tissues.
  • Examined Mosmoa localization at the cellular level in zebrafish and chick embryos.
  • Utilized CRISPR/Cas9 to inactivate both mosmoa and mosmob in zebrafish.

Main Results:

  • Mosmo paralogs are expressed in the head mesenchyme and ventral neural tube of zebrafish embryos.
  • Mosmoa localizes to the plasma membrane, cytoplasmic vesicles, and primary cilium.
  • Inactivation of both mosmo paralogs resulted in frontonasal hypoplasia and craniofacial skeleton defects in adult zebrafish.

Conclusions:

  • Mosmo plays a critical role in vertebrate embryonic development, specifically in craniofacial morphogenesis.
  • MOSMO is a potential candidate gene for unexplained human congenital craniofacial malformations, including those associated with 16p12.1 deletion syndrome.