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

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

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...

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Main Authors: Xiaofei Li, Daogong Zhang, Lei Xu, Wenwen Liu, Na Zhang, Jerome F. Strauss, III, Zhibing Zhang, Haibo Wang
Format: Article
Language:English
Published: Elsevier 2021-12-01
Series:Journal of Pharmacological Sciences
Subjects:
Sperm-associated antigen 6
Vestibular dysfunction
Apoptosis
Hair cells
Scarpa's ganglion cells
Online Access:http://www.sciencedirect.com/science/article/pii/S1347861321000797
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spelling doaj-96b27260a4784c1db9a9cbfff89b56352021-09-03T04:43:56ZengElsevierJournal of Pharmacological Sciences1347-86132021-12-011474325330Sperm-associated antigen 6 (Spag6) mutation leads to vestibular dysfunction in miceXiaofei Li0Daogong Zhang1Lei Xu2Wenwen Liu3Na Zhang4Jerome F. Strauss, III5Zhibing Zhang6Haibo Wang7Department of Otolaryngology-Head and Neck Surgery, Shandong Provincial ENT Hospital, Cheeloo College of Medicine, Shandong University, Jinan, 250022, PR ChinaDepartment of Otolaryngology-Head and Neck Surgery, Shandong Provincial ENT Hospital, Cheeloo College of Medicine, Shandong University, Jinan, 250022, PR ChinaDepartment of Otolaryngology-Head and Neck Surgery, Shandong Provincial ENT Hospital, Cheeloo College of Medicine, Shandong University, Jinan, 250022, PR ChinaDepartment of Otolaryngology-Head and Neck Surgery, Shandong Provincial ENT Hospital, Cheeloo College of Medicine, Shandong University, Jinan, 250022, PR ChinaDepartment of Otolaryngology-Head and Neck Surgery, Shandong Provincial ENT Hospital, Cheeloo College of Medicine, Shandong University, Jinan, 250022, PR ChinaDepartment of Obstetrics and Gynecology, Virginia Commonwealth University, Richmond, VA, 23298, USADepartment of Physiology, Wayne State University, Detroit, MI, 48201, USA; Department of Obstetrics and Gynecology, Wayne State University, Detroit, MI, 48201, USA; Corresponding author. Department of Physiology, Wayne State University, Detroit, MI, 48201, USA.Department of Otolaryngology-Head and Neck Surgery, Shandong Provincial ENT Hospital, Cheeloo College of Medicine, Shandong University, Jinan, 250022, PR China; Corresponding author. Department of Otorhinolaryngology-Head and Neck Surgery, Shandong Provincial ENT Hospital, Cheeloo College of Medicine, Shandong University, Jinan 250022, PR China. Fax: +86 531 83086521.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.http://www.sciencedirect.com/science/article/pii/S1347861321000797Sperm-associated antigen 6Vestibular dysfunctionApoptosisHair cellsScarpa's ganglion cells
collection DOAJ
language English
format Article
sources DOAJ
author Xiaofei Li
Daogong Zhang
Lei Xu
Wenwen Liu
Na Zhang
Jerome F. Strauss, III
Zhibing Zhang
Haibo Wang
spellingShingle Xiaofei Li
Daogong Zhang
Lei Xu
Wenwen Liu
Na Zhang
Jerome F. Strauss, III
Zhibing Zhang
Haibo Wang
Sperm-associated antigen 6 (Spag6) mutation leads to vestibular dysfunction in mice
Journal of Pharmacological Sciences
Sperm-associated antigen 6
Vestibular dysfunction
Apoptosis
Hair cells
Scarpa's ganglion cells
author_facet Xiaofei Li
Daogong Zhang
Lei Xu
Wenwen Liu
Na Zhang
Jerome F. Strauss, III
Zhibing Zhang
Haibo Wang
author_sort Xiaofei Li
title Sperm-associated antigen 6 (Spag6) mutation leads to vestibular dysfunction in mice
title_short Sperm-associated antigen 6 (Spag6) mutation leads to vestibular dysfunction in mice
title_full Sperm-associated antigen 6 (Spag6) mutation leads to vestibular dysfunction in mice
title_fullStr Sperm-associated antigen 6 (Spag6) mutation leads to vestibular dysfunction in mice
title_full_unstemmed Sperm-associated antigen 6 (Spag6) mutation leads to vestibular dysfunction in mice
title_sort sperm-associated antigen 6 (spag6) mutation leads to vestibular dysfunction in mice
publisher Elsevier
series Journal of Pharmacological Sciences
issn 1347-8613
publishDate 2021-12-01
description 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.
topic Sperm-associated antigen 6
Vestibular dysfunction
Apoptosis
Hair cells
Scarpa's ganglion cells
url http://www.sciencedirect.com/science/article/pii/S1347861321000797
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