Sperm-associated antigen 6 (Spag6) mutation leads to vestibular dysfunction in mice.
J Pharmacol Sci
; 147(4): 325-330, 2021 Dec.
Article
em En
| MEDLINE
| ID: mdl-34663514
ABSTRACT
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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Base de dados:
MEDLINE
Assunto principal:
Doenças Vestibulares
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Apoptose
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Proteínas dos Microtúbulos
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Mutação
Idioma:
En
Ano de publicação:
2021
Tipo de documento:
Article