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Updated: Aug 14, 2026

Analyzing Craniofacial Morphogenesis in Zebrafish Using 4D Confocal Microscopy
Published on: January 30, 2014
Inhibition of Jagged-mediated Notch signaling disrupts zebrafish biliary development and generates multi-organ
Kristin Lorent1, Sang-Yeob Yeo, Takaya Oda
1Department of Medicine, University of Pennsylvania School of Medicine, 421 Curie Boulevard, Philadelphia, PA 19104-6058, USA.
Abstract:
The Alagille Syndrome (AGS) is a heritable disorder affecting the liver and other organs. Causative dominant mutations in human Jagged 1 have been identified in most AGS patients. Related organ defects occur in mice that carry jagged 1 and notch 2 mutations. Multiple jagged and notch genes are expressed in the developing zebrafish liver. Compound jagged and notch gene knockdowns alter zebrafish biliary, kidney, pancreatic, cardiac and craniofacial development in a manner compatible with an AGS phenocopy. These data confirm an evolutionarily conserved role for Notch signaling in vertebrate liver development, and support the zebrafish as a model system for diseases of the human biliary system.
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