# Investigating Novel Transcriptional Mechanisms for Visual Neural Circuit Development and Function using Zebrafish

> **NIH NIH P20** · WEST VIRGINIA UNIVERSITY · 2022 · $299,492

## Abstract

Project 003 (486): Investigating Novel Transcriptional Mechanisms for Visual Neural Circuit Development and
Function using Zebrafish, Bergeron, PL
PROJECT SUMMARY / ABSTRACT
Neuroanatomical and behavioral changes associated with neurodevelopmental disorders, such as eye tracking
dysfunction in schizophrenia, are caused by unknown genetic mechanisms. Identifying these critical gene
networks for sensory motor responsiveness in the visual system is one key to developing targeted early
diagnostic and preventative strategies for the symptoms of these disorders that make the world a difficult place
to navigate for those afflicted by them. My long-term goal is to characterize causative genetic mechanisms for
neurodevelopmental disorders-related phenotypes using zebrafish as a model system. This powerful genetic
model provides a unique opportunity as an easily accessible vertebrate with a comparatively “simple” nervous
system, rapid neurodevelopment, many orthologues of human disease-related genes, and defined and tractable
behavioral responses to visual stimuli. We can begin to study specific classes of neurons based on their
molecular identity and function from the moment that they are born in the embryo through adulthood in rapid
time compared to other vertebrate models. My previous work and that of others revealed for the first time the
molecular identity of key interneurons that mediate sensory processing and reflexive control in the mouse and
zebrafish auditory system. These neurons express the transcription factor Genomic Screen Homeobox 1 (Gsx1),
and I sought to determine the role that Gsx1 plays in the development of neural circuits for sensory processing
across the CNS. Upon examination of targeted gsx1 zebrafish mutants, we found profound defects in neuronal
differentiation and axon guidance in the developing visual system. Despite its documented expression in the
developing visual system in mouse and zebrafish, no research has been performed to date to examine the role
that Gsx1 plays in development and function of visual neural circuits. Given the importance of transcriptional
mechanisms for neural circuit development in sensory systems and the proposed ability to target key players in
these large gene networks to treat diseases, we seek to fill this gap in knowledge. Aim 1 of this proposal will
determine the consequences of mutating gsx1 on innate visually mediated behaviors and cell fate determination
in the zebrafish pretectum and the eye. The experiments described in Aim 2 will identify the cellular mechanisms
by which Gsx1 regulates arborization of axons in the pretectum that connect the eyes to the brain. We will be
able to perform these experiments and analyze and validate the large amount of data that will be generated
under the expert guidance of the Visual Function & Morphology Core, as well as the WVU Genomics Core Facility
and the Bioinformatics Core Facility. This research will provide insight into the complex genetic etiology o...

## Key facts

- **NIH application ID:** 10334879
- **Project number:** 1P20GM144230-01
- **Recipient organization:** WEST VIRGINIA UNIVERSITY
- **Principal Investigator:** SADIE A BERGERON
- **Activity code:** P20 (R01, R21, SBIR, etc.)
- **Funding institute:** NIH
- **Fiscal year:** 2022
- **Award amount:** $299,492
- **Award type:** 1
- **Project period:** 2022-03-20 → 2027-01-31

## Primary source

NIH RePORTER: https://reporter.nih.gov/project-details/10334879

## Citation

> US National Institutes of Health, RePORTER application 10334879, Investigating Novel Transcriptional Mechanisms for Visual Neural Circuit Development and Function using Zebrafish (1P20GM144230-01). Retrieved via AI Analytics 2026-05-24 from https://api.ai-analytics.org/grant/nih/10334879. Licensed CC0.

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