# Functions of Fam65b protein complex at the basal stereocilia in hearing and deafness

> **NIH NIH R01** · INDIANA UNIVERSITY INDIANAPOLIS · 2021 · $393,750

## Abstract

PROJECT SUMMARY/ABSTRACT
Hearing loss is a leading cause of morbidity in children affecting about 2 to 3 out of every 1,000 individuals in
the USA. While the study of affected genes has shed important insights into the mechanisms, we are just at
the beginning to understand the disease mechanisms underlying morphogenetic events. The overall goal of
this research is to elucidate the mechanisms that regulate the formation and maintenance of the stereociliary
bundle of cochlear hair cells, and the defects in this process that cause deafness. We propose to investigate
the functions of Fam65b protein complex in the cochlear hair cells and auditory perceptions. In our preliminary
studies, we have identified several components in the Fam65b protein complex. We have generated genomic
modified mouse models using CRISPR/Cas9 system. We have employed immunostaining and illustrated the
stereociliary defects in these mice. Based on our preliminary data, we hypothesize that Fam65b protein
complex is critical for the structural organization of stereociliary bundle of cochlear hair cells. To test our
hypothesis, we will: i) Functionally characterize proteins in the Fam65b protein complex localized at the basal
region of stereocilia; ii) Characterize the biochemical pathways by which Fam65b protein complex regulate hair
cell function and determine the extent to which they modulate the actin cytoskeleton in stereocilia; iii) Continue
our identification of hair cell proteins in the Fam65b protein complex. Our preliminary data show the feasibility
of our approach. We anticipate that our studies will shed new insights into the molecular machinery that
shapes stereocilia and determines its properties. Quite possibly, our findings may link several deafness-related
genes into a common molecular pathway and provide new leads for the development of therapeutic
approaches for the treatment of some forms of the disease.

## Key facts

- **NIH application ID:** 10194456
- **Project number:** 5R01DC017147-04
- **Recipient organization:** INDIANA UNIVERSITY INDIANAPOLIS
- **Principal Investigator:** Bo Zhao
- **Activity code:** R01 (R01, R21, SBIR, etc.)
- **Funding institute:** NIH
- **Fiscal year:** 2021
- **Award amount:** $393,750
- **Award type:** 5
- **Project period:** 2018-07-01 → 2023-06-30

## Primary source

NIH RePORTER: https://reporter.nih.gov/project-details/10194456

## Citation

> US National Institutes of Health, RePORTER application 10194456, Functions of Fam65b protein complex at the basal stereocilia in hearing and deafness (5R01DC017147-04). Retrieved via AI Analytics 2026-05-24 from https://api.ai-analytics.org/grant/nih/10194456. Licensed CC0.

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