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Related Experiment Video

Updated: Mar 26, 2026

4-Dimensional Imaging of Zebrafish Optic Cup Morphogenesis

4-Dimensional Imaging of Zebrafish Optic Cup Morphogenesis

Published on: May 26, 2021

Six3 regulates optic nerve development via multiple mechanisms.

Anat Samuel¹, Ariel M Rubinstein¹, Tehila T Azar¹

¹Department of Medical Neurobiology, Institute for Medical Research - Israel-Canada, The Hebrew University-Hadassah Medical School, Jerusalem, Israel.

Scientific Reports

|January 30, 2016

Summary

This summary is machine-generated.

Transcription factor Six3 is crucial for optic nerve development. Its reduced function in zebrafish causes optic nerve hypoplasia and retinal ganglion cell axon pathfinding errors, revealing new roles in eye and forebrain development.

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Area of Science:

Developmental Biology
Neuroscience
Genetics

Background:

Optic nerve development involves complex axon guidance.
Transcription factors regulating this process are less understood than signaling molecules.
Malformations of the optic nerve can cause vision impairment or blindness.

Purpose of the Study:

Investigate the role of transcription factor Six3 in optic nerve development.
Characterize the effects of reduced Six3 function on retinal ganglion cell (RGC) axon pathfinding.
Elucidate the mechanisms underlying Six3-mediated eye and forebrain malformations.

Main Methods:

Utilized zebrafish as a model organism to study Six3 loss of function.
Analyzed optic nerve hypoplasia and RGC axon pathfinding errors.
Examined eye and optic stalk (OS) patterning and morphogenesis.
Assessed expression of signaling molecules in RGCs and their environment.
Evaluated the diencephalic preoptic area structure.

Main Results:

Reduced Six3 function led to optic nerve hypoplasia and diverse RGC axon pathfinding errors in zebrafish.
Abnormalities were linked to defects in eye and OS development, altered signaling molecule expression, and a deficient diencephalic preoptic area.
Findings highlight Six3's role in regulating eye and forebrain patterning.

Conclusions:

Transcription factor Six3 plays a critical role in multiple aspects of optic nerve development.
Six3 regulates eye and optic stalk morphogenesis and the expression of guidance cues.
The zebrafish Six3 loss-of-function model provides insights into human SIX3-related eye and brain malformations.