ArticleCell and tissue research2008
Lmx1a is required for segregation of sensory epithelia and normal ear histogenesis and morphogenesis.
Article in Cell and tissue research, 2008. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 92 papers.
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Who cites it
92 citing papers in PubMed, 137 citations in OpenAlex.
- A single-nucleus transcriptomic atlas of human inner ear development.Nature neuroscience · 2026Article
- An Lmx1a/b allelic series reveals the role of Lmx1 genes in cochlear nuclei development.Cell and tissue research · 2026Article
- Dicer Deletion in the Ear Can Cut Most Neurons and Their Innervation of Hair Cells to Project to the Ear and the Brainstem.International journal of molecular sciences · 2026Article
- A NovelHuman mutation · 2026Article
- Article
- Sponge bHLH Gene Expression inInternational journal of molecular sciences · 2025Article
- Correlation between the etiology of severe hearing loss and endolymphatic hydrops.European archives of oto-rhino-laryngology : official journal of the European Federation of Oto-Rhino-Laryngological Societies (EUFOS) : affiliated with the German Society for Oto-Rhino-Laryngology - Head and Neck Surgery · 2025Article
- Article
- Development of the semicircular canals and otolithic organs of the vertebrate inner ear.Current topics in developmental biology · 2025Review
- Versican controlled by Lmx1b regulates hyaluronate density and hydration for semicircular canal morphogenesis.Development (Cambridge, England) · 2025Article
- Irx3/5 Null Deletion in Mice Blocks Cochlea-Saccule Segregation and Disrupts the Auditory Tonotopic Map.The Journal of comparative neurology · 2024Article
- Emx2 lineage tracing reveals antecedent patterns of planar polarity in the mouse inner ear.Development (Cambridge, England) · 2024Article
- Versican controlled by Lmx1b regulates hyaluronate density and hydration for semicircular canal morphogenesis.bioRxiv : the preprint server for biology · 2024Article
- Mapping oto-pharyngeal development in a human inner ear organoid model.Development (Cambridge, England) · 2023Article
- Generating high-fidelity cochlear organoids from human pluripotent stem cells.Cell stem cell · 2023Article
- The evolution of the various structures required for hearing inIBRO neuroscience reports · 2023Article
- Regulation of otic neurosensory specification by Notch and Wnt signalling: insights from RNA-seq screenings in the embryonic chicken inner ear.Frontiers in cell and developmental biology · 2023Article
- Molecular mechanisms governing development of the hindbrain choroid plexus and auditory projection: A validation of the seminal observations of Wilhelm His.IBRO neuroscience reports · 2022Review
- SOX9 and SOX10 control fluid homeostasis in the inner ear for hearing through independent and cooperative mechanisms.Proceedings of the National Academy of Sciences of the United States of America · 2022Article
- ISL1 is necessary for auditory neuron development and contributes toward tonotopic organization.Proceedings of the National Academy of Sciences of the United States of America · 2022Article
32 more citing papers are in PubMed but not listed here.
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Authors and funding
6 authors at 3 institutions in 1 country.
Funding
Abstract
At embryonic day 8.5, the LIM-homeodomain factor Lmx1a is expressed throughout the otic placode but becomes developmentally restricted to non-sensory epithelia of the ear (endolymphatic duct, ductus reuniens, cochlea lateral wall). We confirm here that the ears of newborn dreher (Lmx1a (dr)) mutants are dysmorphic. Hair cell markers such as Atoh1 and Myo7 reveal, for the first time, that newborn Lmx1a mutants have only three sensory epithelia: two enlarged canal cristae and one fused epithelium comprising an amalgamation of the cochlea, saccule, and utricle (a "cochlear-gravistatic" endorgan). The enlarged anterior canal crista develops by fusion of horizontal and anterior crista, whereas the posterior crista fuses with an enlarged papilla neglecta that may extend into the cochlear lateral wall. In the fused endorgan, the cochlear region is distinguished from the vestibular region by markers such as Gata3, the presence of a tectorial membrane, and cochlea-specific innervation. The cochlea-like apex displays minor disorganization of the hair and supporting cells. This contrasts with the basal half of the cochlear region, which shows a vestibular epithelium-like organization of hair cells and supporting cells. The dismorphic features of the cochlea are also reflected in altered gene expression patterns. Fgf8 expression expands from inner hair cells in the apex to most hair cells in the base. Two supporting cell marker proteins, Sox2 and Prox1, also differ in their cellular distribution between the base and the apex. Sox2 expression expands in mutant canal cristae prior to their enlargement and fusion and displays a more diffuse and widespread expression in the base of the cochlear region, whereas Prox1 is not detected in the base. These changes in Sox2 and Prox1 expression suggest that Lmx1a expression restricts and sharpens Sox2 expression, thereby defining non-sensory and sensory epithelium. The adult Lmx1a mutant organ of Corti shows a loss of cochlear hair cells, suggesting that the long-term maintenance of hair cells is also disrupted in these mutants.
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