# Integrated multimodal phenotyping of zebrafish models of human cognitive disorders

> **NIH NIH K01** · UTAH STATE HIGHER EDUCATION SYSTEM--UNIVERSITY OF UTAH · 2024 · $162,461

## Abstract

Neurodevelopmental disorders often have clear genetic components but linking genotype to phenotype remains
challenging. Recent advances in genome editing technology have enabled the rapid generation of animal models
for cognitive genetic disorders. However, the differing methodologies used across laboratories to phenotype
these models confounds efforts to make predictive links between cellular functions of the brain and behavioral
outcomes. I propose harnessing Multiplex Intermixed CRISPR droplet (MIC-Drop) technologies to generate
knockouts of all 61 transcriptional regulators present in zebrafish larvae that are associated with human cognitive
disorders. I will characterize these mutants at the molecular and cellular level with a multiplexed single-cell RNA
sequencing technique I have developed, termed MIC-Drop-seq, that allows the simultaneous characterization of
many transcriptomes from hundreds of mutant embryos in a single experiment. I will characterize these mutants
at the brain morphology and behavioral levels with a battery of high throughput quantitative microscopy assays.
I will use these large-scale quantitative datasets to construct new predictive machine learning models that will
uncover the links between genetics, molecular features, brain morphology, and behavioral outcomes in brain
development. These models may yield new treatment strategies for many human cognitive disorders. To achieve
the goals of my proposal, I will embark on an intensive training plan with my mentorship team that will enable
me to transition from my previous background in experimental biology to become a leader in the field of functional
genomics and data informatics. My mentorship team at the University of Utah are leaders in the fields of single-
cell biology, data science, and developmental biology, and will dedicate time to foster my career development
as I transition to lead my own independent research group.

## Key facts

- **NIH application ID:** 10950187
- **Project number:** 1K01HG013682-01
- **Recipient organization:** UTAH STATE HIGHER EDUCATION SYSTEM--UNIVERSITY OF UTAH
- **Principal Investigator:** Clayton Carey
- **Activity code:** K01 (R01, R21, SBIR, etc.)
- **Funding institute:** NIH
- **Fiscal year:** 2024
- **Award amount:** $162,461
- **Award type:** 1
- **Project period:** 2024-09-20 → 2029-08-31

## Primary source

NIH RePORTER: https://reporter.nih.gov/project-details/10950187

## Citation

> US National Institutes of Health, RePORTER application 10950187, Integrated multimodal phenotyping of zebrafish models of human cognitive disorders (1K01HG013682-01). Retrieved via AI Analytics 2026-05-26 from https://api.ai-analytics.org/grant/nih/10950187. Licensed CC0.

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