# Mechanisms of neural compensation in the retina and dysfunction in congenital stationary night blindness

> **NIH NIH F30** · UNIVERSITY OF WISCONSIN-MADISON · 2024 · $40,706

## Abstract

PROJECT SUMMARY/ABSTRACT
The retina is comprised of neural circuit ensembles that communicate through connections called synapses to
generate visual perception and behavior. Retinal diseases cause signaling deficits that derail this communication
and block information flow traveling from the retina to the brain. Severe congenital stationary night blindness is
of particular interest because despite complete suppression of signal transmission through the on retinal pathway
that signals light increments, the on neural circuitry is anatomically intact. Alpha ganglion cells, the primary output
neurons of retinal pathways that code for specific visual features, receive excitatory and inhibitory synaptic
inputs, integrate these inputs across their dendritic compartments, and generate and transmit trains of action
potentials to the brain. We know that neural circuits can adopt diverse strategies to conduct precise synaptic
computations and generate response properties, however, the cellular and synaptic factors prone to alteration
during retinal diseases are not well understood.
This proposal seeks to address important unanswered questions about the mechanisms of neural compensation
that occur in response to specific signaling deficits in well-defined alpha retinal output circuits. Using a set of
neurophysiological and anatomical approaches, these experiments will define how intrinsic properties and
synaptic computations of alpha retinal ganglion cells are altered when the synaptic inputs and balance of
excitation/inhibition that a neuron receives is perturbed. Two CRISPR-edited knockout models of the principal
glutamate receptor of the on retinal pathway, mGluR6, will be used to study neural compensation in the inner
retina across homozygous (100% block) and heterozygous (50% block) conditions. We will correlate single cell
electrophysiology with high resolution imaging and visual behavior assays to complement observations across
the cellular, synaptic, and behavioral levels. Together, the proposed experiments stand to significantly deepen
our mechanistic understanding of the substrates of neural compensation in the inner retina and define the cellular
and synaptic deficits of severe congenital stationary night blindness.

## Key facts

- **NIH application ID:** 10938611
- **Project number:** 5F30EY035128-02
- **Recipient organization:** UNIVERSITY OF WISCONSIN-MADISON
- **Principal Investigator:** Jacob Omar Khoussine
- **Activity code:** F30 (R01, R21, SBIR, etc.)
- **Funding institute:** NIH
- **Fiscal year:** 2024
- **Award amount:** $40,706
- **Award type:** 5
- **Project period:** 2023-07-01 → 2026-06-30

## Primary source

NIH RePORTER: https://reporter.nih.gov/project-details/10938611

## Citation

> US National Institutes of Health, RePORTER application 10938611, Mechanisms of neural compensation in the retina and dysfunction in congenital stationary night blindness (5F30EY035128-02). Retrieved via AI Analytics 2026-05-24 from https://api.ai-analytics.org/grant/nih/10938611. Licensed CC0.

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