ArticleDevelopmental dynamics : an official publication of the American Association of Anatomists2014
The role of Zic genes in inner ear development in the mouse: Exploring mutant mouse phenotypes.
Article in Developmental dynamics : an official publication of the American Association of Anatomists, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers.
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5 citing papers in PubMed, 14 citations in OpenAlex.
- Single-cell transcriptomic profiling of the mouse cochlea: An atlas for targeted therapies.Proceedings of the National Academy of Sciences of the United States of America · 2023Article
- A Model of Waardenburg Syndrome Using Patient-Derived iPSCs With aFrontiers in cell and developmental biology · 2021Article
- The link between inner ear malformations and the rest of the body: what we know so far about genetic, imaging and histology.Neuroradiology · 2020Article
- Sculpting the skull through neurosensory epithelial-mesenchymal signaling.Developmental dynamics : an official publication of the American Association of Anatomists · 2019Review
- Clinicopathological significance of increased ZIC1 expression in human endometrial cancer.Journal of Huazhong University of Science and Technology. Medical sciences = Hua zhong ke ji da xue xue bao. Yi xue Ying De wen ban = Huazhong keji daxue xuebao. Yixue Yingdewen ban · 2015Article
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8 authors at 3 institutions in 2 countries.
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Abstract
backgroundMurine Zic genes (Zic1-5) are expressed in the dorsal hindbrain and in periotic mesenchyme (POM) adjacent to the developing inner ear. Zic genes are involved in developmental signaling pathways in many organ systems, including the ear, although their exact roles haven't been fully elucidated. This report examines the role of Zic1, Zic2, and Zic4 during inner ear development in mouse mutants in which these Zic genes are affected.
resultsZic1/Zic4 double mutants don't exhibit any apparent defects in inner ear morphology. By contrast, inner ears from Zic2(kd/kd) and Zic2(Ku/Ku) mutants have severe but variable morphological defects in endolymphatic duct/sac and semicircular canal formation and in cochlear extension in the inner ear. Analysis of otocyst patterning in the Zic2(Ku/Ku) mutants by in situ hybridization showed changes in the expression patterns of Gbx2 and Pax2.
conclusionsThe experiments provide the first genetic evidence that the Zic genes are required for morphogenesis of the inner ear. Zic2 loss-of-function doesn't prevent initial otocyst patterning but leads to molecular abnormalities concomitant with morphogenesis of the endolymphatic duct. Functional hearing deficits often accompany inner ear dysmorphologies, making Zic2 a novel candidate gene for ongoing efforts to identify the genetic basis of human hearing loss.
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