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Updated: May 11, 2026
09:06
Antibody Uptake Assay for Tracking Notch/Delta Endocytosis During the Asymmetric Division of Zebrafish Radial Glia Progenitors
Published on: January 20, 2023
notch3 is essential for oligodendrocyte development and vascular integrity in zebrafish
Andreas Zaucker1, Sara Mercurio, Nitzan Sternheim
1Department of Developmental and Molecular Biology, Albert Einstein College of Medicine, 1300 Morris Park Avenue, Bronx, NY 10461, USA.
Disease Models & Mechanisms
|May 31, 2013
Summary
Zebrafish notch3 mutations cause vascular problems and affect oligodendrocyte precursor cells, similar to human CADASIL syndrome. These findings highlight Notch3
Area of Science:
- Neuroscience
- Genetics
- Vascular Biology
Background:
- Human NOTCH3 gene mutations cause CADASIL syndrome, a small vessel disease affecting the brain.
- CADASIL is characterized by vasculopathy, neurodegeneration, and dementia.
- Zebrafish offer a model to study NOTCH3 function in vivo.
Purpose of the Study:
- To investigate the role of notch3 in zebrafish development and vascular integrity.
- To understand the cellular and molecular mechanisms underlying notch3-related phenotypes.
- To establish a zebrafish model for CADASIL syndrome research.
Main Methods:
- Generated notch3 mutant zebrafish lines using established screening methods.
- Analyzed oligodendrocyte precursor cell (OPC) development and myelin basic protein (mbp) expression.
- Performed histological and ultrastructural analyses of vascular morphology.
- Assessed gene expression of Notch signaling targets like hey1.
Main Results:
- Zebrafish notch3 mutants exhibited reduced mbp expression and fewer OPCs.
- Adult mutants displayed stress-associated blood accumulation and significant vasculopathy, including vessel dilation and hemorrhage.
- Notch3 deficiency led to reduced hey1 expression, indicating canonical Notch signaling pathway involvement.
- Heterozygous mutants showed altered gene expression, suggesting partial loss-of-function effects.
Conclusions:
- Zebrafish notch3 plays a crucial role in OPC development and mbp expression in larvae.
- Notch3 is essential for maintaining vascular integrity in adult zebrafish.
- Zebrafish notch3 mutants provide a valuable model for studying CADASIL syndrome and Notch3-related vascular disorders.
Related Concept Videos
03:14Notch Signaling Pathway
The Notch signaling pathway is a major intracellular signaling pathway that is highly conserved over a broad spectrum of metazoan species. It stands unique from other intracellular signaling mechanisms in animals because notch protein itself acts as the receptor as well as the primary signaling molecule.
The Notch gene came into the limelight in 1914 after the discovery that its mutation in Drosophila melanogaster leads to a serrated (or "notched") wing margin phenotype. It was not until 1985...
The Notch gene came into the limelight in 1914 after the discovery that its mutation in Drosophila melanogaster leads to a serrated (or "notched") wing margin phenotype. It was not until 1985...
03:14Notch Signaling Pathway
The Notch signaling pathway is a major intracellular signaling pathway that is highly conserved over a broad spectrum of metazoan species. It stands unique from other intracellular signaling mechanisms in animals because notch protein itself acts as the receptor as well as the primary signaling molecule.
The Notch gene came into the limelight in 1914 after the discovery that its mutation in Drosophila melanogaster leads to a serrated (or "notched") wing margin phenotype. It was not until 1985...
The Notch gene came into the limelight in 1914 after the discovery that its mutation in Drosophila melanogaster leads to a serrated (or "notched") wing margin phenotype. It was not until 1985...
01:12Role Of Notch Signalling In Intestinal Stem Cell Renewal
Notch signaling was first discovered in Drosophila melanogaster, where it is involved in cell lineage differentiation. Notch signaling regulates the maintenance and differentiation of intestinal stem cells or ISCs by controlling the expression of atonal homolog 1 or Atoh1. Atoh1 directs cells to differentiate into secretory cells.
Direct cell-to-cell contact is needed for the activation of Notch signaling. The signal is initiated when a notch ligand binds to a receptor on an adjacent cell, also...
Direct cell-to-cell contact is needed for the activation of Notch signaling. The signal is initiated when a notch ligand binds to a receptor on an adjacent cell, also...