Modeling Hearing and Balance Disorders in Mice: The HEar@Jax Workshop - 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, from September 27 to October 2, 2026. This recurring biennial workshop is one of a kind in that it 1) specifically focuses on the use of the mouse model to investigate fundamental and translational questions related to hearing and balance, and 2) offers a compact 5-day format combining morning lectures with afternoon laboratory sessions. The course provides intensive training for 20 participants - graduate or medical students, postdocs or investigators - who are either new to the inner ear field or to mouse models. Six organizers and nine invited speakers, all experts in their respective areas, will give lectures and hands-on training covering a broad spectrum of inner ear research, particularly focused on the peripheral auditory and vestibular systems. Lectures topics will include human and mouse genetics-based gene discovery, mouse models of hearing loss and balance disorders, inner ear development and regeneration, mechanotransduction, neural innervation, brainstem function, noise- and age-related hearing loss, ototoxicity and synaptopathy. Modern techniques such as single cell approaches and viral or CRISPR/Cas9-based gene therapy will also be discussed. An otolaryngologist will provide a clinical perspective on hearing loss patients. Laboratory sessions will include cochlear dissections for immunolabeling or organ culture. Live cultures will be used for calcium imaging, antibiotic-induced damage, and testing of transduction channel function. Participants will use microscopy to examine both fixed and live samples from normal and mutant tissues to observe phenotypes associated with hearing and balance disorders. Additional activities will include gene transfer to the inner ear, preparations of single cells or nuclei for RNA-seq/ATAC-seq, and interactive tutorials for physiological testing (e.g., ABR, DPOAE) and the use of the gEAR database. All lecture slides and protocols will be shared with participants to maximize the long-term value of 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. These informal settings promote networking and collaboration. Participants will also have the opportunity to present a poster on their own research to enhance visibility and engagement. 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), Ulrich Mueller (Johns Hopkins U), Nesrine Benkafadar (Stanford U), Catalina Velez-Ortega (U of Kentucky), Matthew Ingersoll (NIH/NIDCD), Maggie Matern (Merrimack College), Elizabeth Driver (NIH/NIDCD), and Meghan Drummond (Regeneron/CMT foundation).