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https://www.readbyqxmd.com/read/28346477/disruption-of-sorcs2-reveals-differences-in-the-regulation-of-stereociliary-bundle-formation-between-hair-cell-types-in-the-inner-ear
#1
Andrew Forge, Ruth R Taylor, Sally J Dawson, Michael Lovett, Daniel J Jagger
Behavioural anomalies suggesting an inner ear disorder were observed in a colony of transgenic mice. Affected animals were profoundly deaf. Severe hair bundle defects were identified in all outer and inner hair cells (OHC, IHC) in the cochlea and in hair cells of vestibular macular organs, but hair cells in cristae were essentially unaffected. Evidence suggested the disorder was likely due to gene disruption by a randomly inserted transgene construct. Whole-genome sequencing identified interruption of the SorCS2 (Sortilin-related VPS-10 domain containing protein) locus...
March 27, 2017: PLoS Genetics
https://www.readbyqxmd.com/read/28343884/short-term-desensitization-of-fast-escape-behavior-associated-with-suppression-of-mauthner-cell-activity-in-larval-zebrafish
#2
Megumi Takahashi, Maya Inoue, Masashi Tanimoto, Tsunehiko Kohashi, Yoichi Oda
Escape is among the simplest animal behaviors employed to study the neural mechanisms underlying learning. Teleost fishes exhibit behavioral learning of fast escape initiated with a C-shaped body bend (C-start). C-starts are subdivided into short-latency (SLC) and long-latency (LLC) types in larval zebrafish. Whether these two can be separately modified, and the neural correlates of this modification, however, remains undetermined. We thus performed Ca(2+) imaging of Mauthner (M-) cells, a pair of giant hindbrain neurons constituting a core element of SLC circuit, during behavioral learning in larval zebrafish...
March 23, 2017: Neuroscience Research
https://www.readbyqxmd.com/read/28332011/delayed-otolith-development-does-not-impair-vestibular-circuit-formation-in-zebrafish
#3
Richard Roberts, Jeffrey Elsner, Martha W Bagnall
What is the role of normally patterned sensory signaling in development of vestibular circuits? For technical reasons, including the difficulty in depriving animals of vestibular inputs, this has been a challenging question to address. Here we take advantage of a vestibular-deficient zebrafish mutant, rock solo (AN66) , in order to examine whether normal sensory input is required for formation of vestibular-driven postural circuitry. We show that the rock solo (AN66) mutant is a splice site mutation in the secreted glycoprotein otogelin (otog), which we confirm through both whole genome sequencing and complementation with an otog early termination mutant...
March 22, 2017: Journal of the Association for Research in Otolaryngology: JARO
https://www.readbyqxmd.com/read/28292428/cell-cell-contact-area-affects-notch-signaling-and-notch-dependent-patterning
#4
Oren Shaya, Udi Binshtok, Micha Hersch, Dmitri Rivkin, Sheila Weinreb, Liat Amir-Zilberstein, Bassma Khamaisi, Olya Oppenheim, Ravi A Desai, Richard J Goodyear, Guy P Richardson, Christopher S Chen, David Sprinzak
During development, cells undergo dramatic changes in their morphology. By affecting contact geometry, these morphological changes could influence cellular communication. However, it has remained unclear whether and how signaling depends on contact geometry. This question is particularly relevant for Notch signaling, which coordinates neighboring cell fates through direct cell-cell signaling. Using micropatterning with a receptor trans-endocytosis assay, we show that signaling between pairs of cells correlates with their contact area...
March 13, 2017: Developmental Cell
https://www.readbyqxmd.com/read/28271491/role-of-skeletal-muscle-in-ear-development
#5
REVIEW
Irena Rot, Mark Baguma-Nibasheka, Willard J Costain, Paul Hong, Robert Tafra, Snjezana Mardesic-Brakus, Natasa Mrduljas-Djujic, Mirna Saraga-Babic, Boris Kablar
The current paper is a continuation of our work described in Rot and Kablar, 2010. Here, we show lists of 10 up- and 87 down-regulated genes obtained by a cDNA microarray analysis that compared developing Myf5-/-:Myod-/- (and Mrf4-/-) petrous part of the temporal bone, containing middle and inner ear, to the control, at embryonic day 18.5. Myf5-/-:Myod-/- fetuses entirely lack skeletal myoblasts and muscles. They are unable to move their head, which interferes with the perception of angular acceleration. Previously, we showed that the inner ear areas most affected in Myf5-/-:Myod-/- fetuses were the vestibular cristae ampullaris, sensitive to angular acceleration...
March 8, 2017: Histology and Histopathology
https://www.readbyqxmd.com/read/28266911/transcription-factor-emx2-controls-stereociliary-bundle-orientation-of-sensory-hair-cells
#6
Tao Jiang, Katie Kindt, Doris K Wu
The asymmetric location of stereociliary bundle (hair bundle) on apical surface of mechanosensory hair cells (HCs) dictates the direction in which a given HC can respond to cues such as sound, head movements, and water pressure. Notably, vestibular sensory organs of the inner ear, the maculae, exhibit a line of polarity reversal (LPR) across which, hair bundles are polarized in a mirror-image pattern. Similarly, HCs in neuromasts of the zebrafish lateral line system are generated as pairs, and two sibling HCs develop opposite hair bundle orientations...
March 7, 2017: ELife
https://www.readbyqxmd.com/read/28264554/hearing-improvement-in-a-j-mice-via-the-mouse-nerve-growth-factor
#7
Lixiang Gao, Ruli Ge, Gang Xie, Dandan Yao, Ping Li, Oumei Wang, Xiufang Ma, Fengchan Han
Objectives: To investigate the otoprotective effects of mouse nerve growth factor (mNGF) in A/J mice. Methods: The mice at postnatal day 7 (P7) were randomly separated into a mNGF treated group (mNGF group) and a distilled water (for injection) treated group (control group). The mNGF dissolved in distilled water or distilled water alone was given to the mice once every other day from P7 by intramuscular injection in the hips. The otoprotective effects of mNGF in A/J mice were observed in a time course manner...
March 8, 2017: Clinical and Experimental Otorhinolaryngology
https://www.readbyqxmd.com/read/28263708/supporting-cells-remove-and-replace-sensory-receptor-hair-cells-in-a-balance-organ-of-adult-mice
#8
Stephanie A Bucks, Brandon C Cox, Brittany A Vlosich, James P Manning, Tot B Nguyen, Jennifer S Stone
Vestibular hair cells in the inner ear encode head movements and mediate the sense of balance. These cells undergo cell death and replacement (turnover) throughout life in non-mammalian vertebrates. However, there is no definitive evidence that this process occurs in mammals. We used fate-mapping and other methods to demonstrate that utricular type II vestibular hair cells undergo turnover in adult mice under normal conditions. We found that supporting cells phagocytose both type I and II hair cells. Plp1-CreER(T2)-expressing supporting cells replace type II hair cells...
March 6, 2017: ELife
https://www.readbyqxmd.com/read/28259711/allicin-protects-against-cisplatin-induced-vestibular-dysfunction-by-inhibiting-the-apoptotic-pathway
#9
Xianmin Wu, Jing Cai, Xiaofei Li, He Li, Jianfeng Li, Xiaohui Bai, Wenwen Liu, Yuechen Han, Lei Xu, Daogong Zhang, Haibo Wang, Zhaomin Fan
Cisplatin is an anticancer drug that causes the impairment of inner ear function as side effects, including hearing loss and balance dysfunction. The purpose of this study was to investigate the effects of allicin against cisplatin-induced vestibular dysfunction in mice and to make clear the mechanism underlying the protective effects of allicin on oto-vestibulotoxicity. Mice intraperitoneally injected with cisplatin exhibited vestibular dysfunction in swimming test, which agreed with impairment in vestibule...
March 1, 2017: European Journal of Pharmacology
https://www.readbyqxmd.com/read/28254438/gene-therapy-restores-balance-and-auditory-functions-in-a-mouse-model-of-usher-syndrome
#10
Kevin Isgrig, Jack W Shteamer, Inna A Belyantseva, Meghan C Drummond, Tracy S Fitzgerald, Sarath Vijayakumar, Sherri M Jones, Andrew J Griffith, Thomas B Friedman, Lisa L Cunningham, Wade W Chien
Dizziness and hearing loss are among the most common disabilities. Many forms of hereditary balance and hearing disorders are caused by abnormal development of stereocilia, mechanosensory organelles on the apical surface of hair cells in the inner ear. The deaf whirler mouse, a model of human Usher syndrome (manifested by hearing loss, dizziness, and blindness), has a recessive mutation in the whirlin gene, which renders hair cell stereocilia short and dysfunctional. In this study, wild-type whirlin cDNA was delivered to the inner ears of neonatal whirler mice using adeno-associated virus serotype 2/8 (AAV8-whirlin) by injection into the posterior semicircular canal...
March 1, 2017: Molecular Therapy: the Journal of the American Society of Gene Therapy
https://www.readbyqxmd.com/read/28245737/hearing-disorders-in-cats
#11
George M Strain
Practical relevance: Auditory function is a sense that is central to life for cats - being important in situational awareness of potential predators, pursuit of prey, and for communication with conspecifics, humans and other species. Deafness in cats is most frequently the result of a genetic disorder, strongly associated with white fur and blue eyes, but may also result from acquired causes such as advancing age, ototoxic drugs, infection, environmental noise and physical trauma. Deafness can be sensorineural, where there is loss of cochlear hair cells, or conductive, where sound is muffled on its way to the inner ear...
March 2017: Journal of Feline Medicine and Surgery
https://www.readbyqxmd.com/read/28230212/establishment-of-planar-cell-polarity-is-coupled-to-regional-cell-cycle-exit-and-cell-differentiation-in-the-mouse-utricle
#12
Xiaoyu Yang, Xiaoqing Qian, Rui Ma, Xinwei Wang, Juanmei Yang, Wenwei Luo, Ping Chen, Fanglu Chi, Dongdong Ren
Sensory hair cells are coordinately oriented within each inner ear sensory organ to exhibit a particular form of planar cell polarity (PCP) necessary for mechanotransduction. However, the developmental events associated with establishing PCP in the vestibule are unclear, hindering data interpretation and employment of the vestibule for PCP studies. Herein, we investigated PCP of the mouse vestibular organs. We further characterised cell cycle exit, cell differentiation, and PCP establishment in the utricle...
February 23, 2017: Scientific Reports
https://www.readbyqxmd.com/read/28228581/spaceflight-induced-synaptic-modifications-within-hair-cells-of-the-mammalian-utricle
#13
David R Sultemeier, Kristel R Choy, Felix E Schweizer, Larry F Hoffman
Exposure to the microgravity conditions of spaceflight alleviates the load normally imposed by the Earth's gravitational field upon the inner ear utricular epithelia. Previous ultrastructural investigations showed that spaceflight induced an increase in synapse density within hair cells of the rat utricle. However, the utricle exhibits broad physiologic heterogeneity across different epithelial regions, and it is unknown whether capabilities for synaptic plasticity generalize to hair cells across its topography...
February 22, 2017: Journal of Neurophysiology
https://www.readbyqxmd.com/read/28224282/micrornas-effective-elements-in-ear-related-diseases-and-hearing-loss
#14
REVIEW
Mohammad-Reza Mahmoudian-Sani, Ameneh Mehri-Ghahfarrokhi, Fereshteh Ahmadinejad, Morteza Hashemzadeh-Chaleshtori, Massoud Saidijam, Mohammad-Saeid Jami
miRNAs are important factors for post-transcriptional process that controls gene expression at mRNA level. Various biological processes, including growth and differentiation, are regulated by miRNAs. miRNAs have been demonstrated to play an essential role in development and progression of hearing loss. Nowadays, miRNAs are known as critical factors involved in different physiological, biological, and pathological processes, such as gene expression, progressive sensorineural hearing loss, age-related hearing loss, noise-induced hearing loss, cholesteatoma, schwannomas, and inner ear inflammation...
February 21, 2017: European Archives of Oto-rhino-laryngology
https://www.readbyqxmd.com/read/28223157/dose-dependent-cochlear-and-vestibular-toxicity-of-trans-tympanic-cisplatin-in-the-rat
#15
Angela Callejo, Amandine Durochat, Stéphanie Bressieux, Aurélie Saleur, Christian Chabbert, Ivan Domènech Juan, Jordi Llorens, Sophie Gaboyard-Niay
In vivo studies are needed to study cisplatin ototoxicity and to evaluate candidate protective treatments. Rats and mice are the preferred species for toxicological and pharmacological pre-clinical research, but systemic administration of cisplatin causes high morbidity in these species. We hypothesized that trans-tympanic administration of cisplatin would provide a good model for studying its auditory and vestibular toxicity in the rat. Cisplatin was administered by the trans-tympanic route in one ear (50μl, 0...
February 14, 2017: Neurotoxicology
https://www.readbyqxmd.com/read/28220837/a-novel-role-of-the-organizer-gene-goosecoid-as-an-inhibitor-of-wnt-pcp-mediated-convergent-extension-in-xenopus-and-mouse
#16
Bärbel Ulmer, Melanie Tingler, Sabrina Kurz, Markus Maerker, Philipp Andre, Dina Mönch, Marina Campione, Kirsten Deißler, Mark Lewandoski, Thomas Thumberger, Axel Schweickert, Abraham Fainsod, Herbert Steinbeißer, Martin Blum
Goosecoid (Gsc) expression marks the primary embryonic organizer in vertebrates and beyond. While functions have been assigned during later embryogenesis, the role of Gsc in the organizer has remained enigmatic. Using conditional gain-of-function approaches in Xenopus and mouse to maintain Gsc expression in the organizer and along the axial midline, neural tube closure defects (NTDs) arose and dorsal extension was compromised. Both phenotypes represent convergent extension (CE) defects, arising from impaired Wnt/planar cell polarity (PCP) signaling...
February 21, 2017: Scientific Reports
https://www.readbyqxmd.com/read/28196606/imaging-and-cell-count-in-cleared-intact-cochlea-in-the-mongolian-gerbil-using-laser-scanning-confocal-microscopy
#17
M Risoud, J Sircoglou, G Dedieu, M Tardivel, C Vincent, N-X Bonne
OBJECTIVES: To draw up a clearing protocol for Mongolian gerbil cochlea, and to assess the feasibility of quantifying and analyzing 3D cell architecture in the transparent cochleae. MATERIALS AND METHODS: Freshly dissected inner ears were prepared on a 13-day protocol: fixation, microdissection, post-fixation, decalcification, pretreatment (signal enhancement, permeabilization and blocking), fluorescent labeling (indirect immunolabeling and direct labeling), dehydration, clearing in Spalteholz solution (MSBB: methyl salicylate and benzyl benzoate) and mounting...
February 9, 2017: European Annals of Otorhinolaryngology, Head and Neck Diseases
https://www.readbyqxmd.com/read/28181574/indispensable-role-of-mdm2-p53-interaction-during-the-embryonic-and-postnatal-inner-ear-development
#18
M Laos, M Sulg, A Herranen, T Anttonen, U Pirvola
p53 is a key component of a signaling network that protects cells against various stresses. As excess p53 is detrimental to cells, its levels are tightly controlled by several mechanisms. The E3 ubiquitin ligase Mdm2 is a major negative regulator of p53. The significance of balanced p53 levels in normal tissues, at different stages of lifetime, is poorly understood. We have studied in vivo how the disruption of Mdm2/p53 interaction affects the early-embryonic otic progenitor cells and their descendants, the auditory supporting cells and hair cells...
February 9, 2017: Scientific Reports
https://www.readbyqxmd.com/read/28181545/gfi1-cre-mice-have-early-onset-progressive-hearing-loss-and-induce-recombination-in-numerous-inner-ear-non-hair-cells
#19
Maggie Matern, Sarath Vijayakumar, Zachary Margulies, Beatrice Milon, Yang Song, Ran Elkon, Xiaoyu Zhang, Sherri M Jones, Ronna Hertzano
Studies of developmental and functional biology largely rely on conditional expression of genes in a cell type-specific manner. Therefore, the importance of specificity and lack of inherent phenotypes for Cre-driver animals cannot be overemphasized. The Gfi1(Cre) mouse is commonly used for conditional hair cell-specific gene deletion/reporter gene activation in the inner ear. Here, using immunofluorescence and flow cytometry, we show that the Gfi1(Cre) mice produce a pattern of recombination that is not strictly limited to hair cells within the inner ear...
February 9, 2017: Scientific Reports
https://www.readbyqxmd.com/read/28179572/spectrin-%C3%AE-v-adaptive-mutations-and-changes-in-subcellular-location-correlate-with-emergence-of-hair-cell-electromotility-in-mammalians
#20
Matteo Cortese, Samantha Papal, Francisco Pisciottano, Ana Belén Elgoyhen, Jean-Pierre Hardelin, Christine Petit, Lucia Florencia Franchini, Aziz El-Amraoui
The remarkable hearing capacities of mammals arise from various evolutionary innovations. These include the cochlear outer hair cells and their singular feature, somatic electromotility, i.e., the ability of their cylindrical cell body to shorten and elongate upon cell depolarization and hyperpolarization, respectively. To shed light on the processes underlying the emergence of electromotility, we focused on the βV giant spectrin, a major component of the outer hair cells' cortical cytoskeleton. We identified strong signatures of adaptive evolution at multiple sites along the spectrin-βV amino acid sequence in the lineage leading to mammals, together with substantial differences in the subcellular location of this protein between the frog and the mouse inner ear hair cells...
February 8, 2017: Proceedings of the National Academy of Sciences of the United States of America
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