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Publication : Sperm-associated antigen 6 (Spag6) mutation leads to vestibular dysfunction in mice.

First Author  Li X Year  2021
Journal  J Pharmacol Sci Volume  147
Issue  4 Pages  325-330
PubMed ID  34663514 Mgi Jnum  J:322360
Mgi Id  MGI:7258214 Doi  10.1016/j.jphs.2021.08.004
Citation  Li X, et al. (2021) Sperm-associated antigen 6 (Spag6) mutation leads to vestibular dysfunction in mice. J Pharmacol Sci 147(4):325-330
abstractText  Spag6 encodes an axoneme central apparatus protein that is required for normal flagellar and cilia motility. Recent findings suggest that Spag6 plays a role in hearing and planar cell polarity (PCP) in the cochlea of the inner ear. However, a role for Spag6 in the vestibule has not yet been explored. In the present study, the function of Spag6 in the vestibule of the inner ear was examined using Spag6-deficient mice. Our results demonstrate a vestibular disorder in the Spag6 mutants, associated with abnormal ultrastructures of vestibular hair cells and Scarpa's ganglion cells, including swollen stereocilia, decreased crista in mitochondria and swollen Scarpa's ganglion cells. Immunostaining data suggests existence of caspase-dependent apoptosis in vestibular sensory epithelium and Scarpa's ganglion cells. Our observations reveal new functions for Spag6 in vestibular function and apoptosis in the mouse vestibule.
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