PROJECT SUMMARY The funds requested in this application are for partial support of the ?Modeling Hearing and Balance Disorders in Mice: The HEar@JAX Workshop?, to be held at The Jackson Laboratory (JAX) in Bar Harbor, Maine, September 23-28, 2018. This hands-on workshop is one of a kind as it focuses on mouse models of human auditory and vestibular disorders. It represents an intensive training opportunity for 20 graduate students, postdocs and investigators wishing to gain expertise in the biology and functional characterization of the mouse auditory and vestibular systems. The course is structured with classroom lectures in the morning, followed by hands-on laboratory sessions and tutorials in the afternoon. Evening lectures will feature plenary presentations by senior researchers in the field. Participants will also have the opportunity to present their own research and there will be dedicated time for discussions of potential career opportunities. Six members of the organizing committee, three Jackson Laboratory speakers and ten invited faculty who all committed to participate pending funding will give the lectures and the laboratory training. During hands-on laboratory sessions, participants will learn how to dissect the cochlea and the vestibular organs and perform whole-mount immunofluorescence stainings using typical antibodies to label hair cells, supporting cells and neuron projections. The sensory organs will also be placed in culture to perform dye uptake and show aminoglycoside toxicity in hair cells. Cochlear and vestibular samples collected by participants will be mounted and observed using confocal microscopy. Mutant samples prepared in advance will enable participants to observe phenotypes leading to congenital hearing loss and circling behavior. Finally, tutorials and demonstrations will be presented on various topics including inner ear paint-fill, physiology (Auditory brainstem response and Distortion product otoacoustic emission recordings) and therapy (Organoids, Inner ear gene transfer). One of the hallmarks of this workshop is the opportunity for participants to closely interact with the faculty at the Highseas Conference Center, which also serves as the residence and dining facility for attendees. Having meals together as well as other social activities encourages network development and fosters collaboration. Invited speakers: Uli Muller (Johns Hopkins), Karen Steel (King's college), Neil Segil (USC), Ronna Hertzano (U of Maryland), Uri Manor (Salk Institute), Jennifer Stone (U of Washington), Paul Fuchs (Johns Hopkins), Andy Groves (Baylor), Gwen Geleoc (Boston Children's hospital), Karl Koehler (Indiana U). Organizers: Karen Avraham (Tel-Aviv U), Matt Kelley (NIH), Guy Richardson (U of Sussex), Amanda Lauer (Johns Hopkins), Mike Bowl (MRC Harwell), Basile Tarchini (The Jackson Laboratory).