PROJECT SUMMARY This application seeks partial support for “Modeling Hearing and Balance Disorders in Mice: The HEar@JAX Workshop”, to be held at The Jackson Laboratory (JAX) in Bar Harbor, ME, on September 15-20, 2024. This recurring biennial workshop is one of a kind as it 1) specifically focuses on the use of the mouse model to investigate fundamental and translational questions about hearing and balance, and 2) has a compact 5-day format with morning lectures and afternoon laboratory time. The course is an intensive training opportunity for 20 graduate or medical students, postdocs or investigators who are either new to the inner ear field, or new to the mouse model. Six organizers and eight invited speakers are ideally qualified to give lectures and laboratory training in their own fields to broadly cover inner ear research, in particular the periphery. The lectures will address human and mouse genetics-based gene discovery, mouse models of hearing loss and balance disorders, development and regeneration of the inner ear, mechanotransduction, afferent and efferent innervation, brainstem function, noise- and age-related hearing loss, ototoxicity and synaptopathy. Modern techniques including single cell approaches and viral and CRISPR/Cas9-based gene therapy will be discussed. An otolaryngologist’s perspective on patients with hearing loss will also be presented. Laboratory time will include cochlear dissections for immunolabeling or for organ culture. Live cultures will be used for calcium imaging, antibiotic damage and to test transduction channel function. Confocal microscopy of fixed or live samples representing normal and mutant tissues will enable participants to observe defects resulting in hearing loss and vestibular disorders. Other activities will include inner ear gene transfer, paint-fill, as well as practical and interactive tutorials for physiological testing (e.g., ABR, DPOAE) and to navigate the gEAR database. All lecture slides and protocols introduced during the course will be made available to participants to maximize the benefits of attending the course. A hallmark of this workshop is the close interaction between participants and faculty at the Highseas Conference Center, which also serves as the residence and dining facility. Sharing meals and other social activities encourages network development and fosters collaboration. To enhance their visibility, participants will have the opportunity to present a poster about their own research. Course organizers: Matt Kelley (NIH/NIDCD; Chair), Guy Richardson (U of Sussex), Ronna Hertzano (U of Maryland, NIH/NIDCD), Cat Weisz (NIH/NIDCD), Mike Bowl (U College London), Basile Tarchini (The Jackson Laboratory, R13 PI). Invited speakers: Radha Kalluri (U of Southern California), Hela Azaiez (U of Iowa), Gwenaelle Geleoc (Harvard U), Ulrich Mueller (Johns Hopkins U), Lisa Olson (Columbia U), Betsy Driver (NIH/NIDCD), Vidhya Munnamalai (The Jackson Laboratory), Ksenia Gnedeva (U of Sou...