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Harmonin (Ush1c) is required in zebrafish Muller glial cells for photoreceptor synaptic development and function

Phillips, Jennifer B; Blanco-Sanchez, Bernardo; Lentz, Jennifer J; Tallafuss, Alexandra; Khanobdee, Kornnika; Sampath, Srirangan; Jacobs, Zachary G; Han, Philip F; Mishra, Monalisa; Titus, Tom A; Williams, David S; Keats, Bronya; Washbourne, Philip; Westerfield, Monte


Usher syndrome is the most prevalent cause of hereditary deaf-blindness, characterized by congenital sensorineural hearing impairment and progressive photoreceptor degeneration beginning in childhood or adolescence. Diagnosis and management of this disease are complex, and the molecular changes underlying sensory cell impairment remain poorly understood. Here we characterize two zebrafish models for a severe form of Usher syndrome, Usher syndrome type 1C (USH1C): one model is a mutant with a...[Show more]

CollectionsANU Research Publications
Date published: 2011
Type: Journal article
Source: Disease Models and Mechanisms
DOI: 10.1242/dmm.006429


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