ArticleHuman molecular genetics2013
GATA3 controls the specification of prosensory domain and neuronal survival in the mouse cochlea.
Article in Human molecular genetics, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 51 papers.
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51 citing papers in PubMed, 78 citations in OpenAlex.
- Hypomagnesemia in Hypoparathyroidism, Sensorineural Deafness, and Renal Dysplasia Syndrome Caused by Pathogenic Variants ofKidney international reports · 2026Article
- A single-nucleus transcriptomic atlas of human inner ear development.Nature neuroscience · 2026Article
- Genetic Findings in Seven Cochlear Implanted Patients with Severe-to-Profound Hearing Loss.Genes · 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
- Kolliker's Organ and Its Functional Role in the Development of Corti's Organ and Auditory Systems.Audiology research · 2025Review
- A primate model animal revealed the inter-species differences and similarities in the subtype specifications of the spiral ganglion neurons.Scientific reports · 2024Article
- A novelMolecular genetics and metabolism reports · 2024Article
- ISL1 and POU4F1 Directly Interact to Regulate the Differentiation and Survival of Inner Ear Sensory Neurons.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2024Article
- Shared features in ear and kidney development - implications for oto-renal syndromes.Disease models & mechanisms · 2024Article
- [Research progress on hereditary endocrine and metabolic diseases associated with sensorineural hearing loss].Lin chuang er bi yan hou tou jing wai ke za zhi = Journal of clinical otorhinolaryngology head and neck surgery · 2024Article
- Uncovering the role of FOXA2 in the Development of Human Serotonin Neurons.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2023Article
- Human pluripotent stem cell-derived inner ear organoids recapitulate otic development in vitro.Development (Cambridge, England) · 2023Article
- Gata3 is required in late proneurosensory development for proper sensory cell formation and organization.Scientific reports · 2023Article
- Generating high-fidelity cochlear organoids from human pluripotent stem cells.Cell stem cell · 2023Article
- A single-cell level comparison of human inner ear organoids with the human cochlea and vestibular organs.Cell reports · 2023Article
- Gata3 is Required in Late Proneurosensory Development for Proper Sensory Cell Formation and Organization.Research square · 2023Article
- Tbx2 and Tbx3 regulate cell fate progression of the otic vesicle for inner ear development.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
- Sufu- and Spop-mediated regulation of Gli2 is essential for the control of mammalian cochlear hair cell differentiation.Proceedings of the National Academy of Sciences of the United States of America · 2022Article
- Early development of the cochlea of the common marmoset, a non-human primate model.Neural development · 2022Article
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Authors and funding
11 authors at 5 institutions in 2 countries.
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Abstract
HDR syndrome (also known as Barakat syndrome) is a developmental disorder characterized by hypoparathyroidism, sensorineural deafness and renal disease. Although genetic mapping and subsequent functional studies indicate that GATA3 haplo-insufficiency causes human HDR syndrome, the role of Gata3 in sensorineural deafness and auditory system development is largely unknown. In this study, we show that Gata3 is continuously expressed in the developing mouse inner ear. Conditional knockout of Gata3 in the developing inner ear disrupts the morphogenesis of mouse inner ear, resulting in a disorganized and shortened cochlear duct with significant fewer hair cells and supporting cells. Loss of Gata3 function leads to the failure in the specification of prosensory domain and subsequently, to increased cell death in the cochlear duct. Moreover, though the initial generation of cochleovestibular ganglion (CVG) cells is not affected in Gata3-null mice, spiral ganglion neurons (SGNs) are nearly depleted due to apoptosis. Our results demonstrate the essential role of Gata3 in specifying the prosensory domain in the cochlea and in regulating the survival of SGNs, thus identifying a molecular mechanism underlying human HDR syndrome.
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