In a large scale screen for genetic defects in zebrafish embryogenesis we identified mutations affecting several aspects of ear development, including: specification of the otic placode, growth of the otic vesicle (otocyst), otolith formation, morphogenesis of the semicircular canals and differentiation of the otic capsule. Here we report initial phenotypic and genetic characterization of 20 of these mutations defining 13 independent loci. Embryos mutant at the quadro locus display abnormal specification of the otic placode. As revealed by dlx-3 expression, the otic field in the mutant embryos is smaller or split into two fields. At later stages of development the ear of quadro mutants is frequently divided into two smaller, incomplete units. Four loci affect ear shape shortly after formation of the otic vesicle. All of them also display abnormal brain morphology. Mutations in five loci result in the absence of otolith formation; two of these also produce changes of ear morphology. Two loci, little richard and golas, affect morphology of the otic vesicle shortly before formation of the semicircular canals. In both cases the morphogenesis of the semicircular canals is disrupted. Finally, the antytalent locus is involved in late expansion of the ear structure. Analysis of mutations presented here will strengthen our understanding of vertebrate ear morphogenesis and provide novel entry points to its genetic analysis.
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http://dx.doi.org/10.1242/dev.123.1.275 | DOI Listing |
PLoS Pathog
January 2025
School of Clinical Dentistry, University of Sheffield, Sheffield, United Kingdom.
Porphyromonas gingivalis (Pg) is a keystone pathogen in periodontitis, a highly prevalent disease manifested by chronic inflammation of the periodontium, alveolar bone resorption and tooth loss. During periodontitis pathobionts such as Pg can enter the bloodstream and growing evidence correlates periodontitis with increased risk of cardiovascular and neurodegenerative diseases. However, the mechanism by which immune cells respond to Pg challenge in vivo remains elusive.
View Article and Find Full Text PDFChemosphere
February 2025
Department of Life Science, School of Life Science, National Taiwan Normal University, Taipei, Taiwan. Electronic address:
Acidification of freshwater due to human activities is a widespread environmental problem. Its effects on the sensorimotor responses of fish, particularly during embryonic stages, may affect population fitness. To address this, zebrafish embryos were exposed to water at pH 7, 5 and 4.
View Article and Find Full Text PDFJ Vis Exp
November 2024
Department of Otolaryngology - Head & Neck Surgery, Johns Hopkins University School of Medicine.
The living human inner ear is challenging to study because it is encased within dense otic capsule bone that limits access to biological tissue. Traditional temporal bone histopathology methods rely on lengthy, expensive decalcification protocols that take 9-10 months and reduce the types of tissue analysis possible due to RNA degradation. There is a critical need to develop methods to access fresh human inner ear tissue to better understand otologic diseases, such as Ménière's disease, at the cellular and molecular level.
View Article and Find Full Text PDFDevelopment
December 2024
Department of Medicine and Life Sciences, Universitat Pompeu Fabra - Parc de Recerca Biomèdica de Barcelona, Carrer del Doctor Aiguader 8808003 Barcelona, Spain.
Human inner ear organoids are three-dimensional tissular structures grown in vitro that recapitulate some aspects of the fetal inner ear and allow the differentiation of inner ear cell types. These organoids offer a system in which to study human inner ear development, mutations causing hearing loss and vertigo, and new therapeutic drugs. However, the extent to which such organoids mimic in vivo human inner ear development and cellular composition remains unclear.
View Article and Find Full Text PDFInt J Mol Sci
October 2024
Nantong Laboratory of Development and Diseases, School of Life Sciences, Co-Innovation Center of Neuroregeneration, Nantong University, Qixiu Road 19, Nantong 226001, China.
The development of sensory hair cells (HCs) is closely linked to hearing loss. There are still many unidentified genes that may play a crucial role in HC development and function. Glutamine synthetase, Glul, is expressed in sensory hair cells and auditory organs.
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