ArticleBMC developmental biology2009
Tbx1 and Brn4 regulate retinoic acid metabolic genes during cochlear morphogenesis.
Article in BMC developmental biology, 2009. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers.
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26 citing papers in PubMed, 49 citations in OpenAlex.
- Primary and secondary defects of the thymus.Immunological reviews · 2024Review
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- Chromatin remodeler CHD7 is critical for cochlear morphogenesis and neurosensory patterning.Developmental biology · 2021Article
- Pharmacological Rescue of the Brain Cortex Phenotype ofFrontiers in molecular neuroscience · 2021Article
- TBX1 is required for normal stria vascularis and semicircular canal development.Developmental biology · 2020Article
- Sculpting the skull through neurosensory epithelial-mesenchymal signaling.Developmental dynamics : an official publication of the American Association of Anatomists · 2019Review
- Anatomic Malformations of the Middle and Inner Ear in 22q11.2 Deletion Syndrome: Case Series and Literature Review.AJNR. American journal of neuroradiology · 2018Review
- Article
- Otologic and audiologic findings in 22q11.2 deletion syndrome.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 · 2017Article
- QTL Mapping of Endocochlear Potential Differences between C57BL/6J and BALB/cJ mice.Journal of the Association for Research in Otolaryngology : JARO · 2016Article
- Modeling a model: Mouse genetics, 22q11.2 Deletion Syndrome, and disorders of cortical circuit development.Progress in neurobiology · 2015Review
- Article
- The role of Zic genes in inner ear development in the mouse: Exploring mutant mouse phenotypes.Developmental dynamics : an official publication of the American Association of Anatomists · 2014Article
- Dysphagia and disrupted cranial nerve development in a mouse model of DiGeorge (22q11) deletion syndrome.Disease models & mechanisms · 2014Article
- Immunodeficiency in DiGeorge Syndrome and Options for Treating Cases with Complete Athymia.Frontiers in immunology · 2013Review
- Chimeric negative regulation of p14ARF and TBX1 by a t(9;22) translocation associated with melanoma, deafness, and DNA repair deficiency.Human mutation · 2013Article
- 22q11 Gene dosage establishes an adaptive range for sonic hedgehog and retinoic acid signaling during early development.Human molecular genetics · 2013Article
- Article
- Molecular mechanisms of inner ear development.Cold Spring Harbor perspectives in biology · 2012Review
- Identification of putative retinoic acid target genes downstream of mesenchymal Tbx1 during inner ear development.Developmental dynamics : an official publication of the American Association of Anatomists · 2012Article
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Authors and funding
5 authors at 1 institution in 1 country.
Funding
Abstract
backgroundIn vertebrates, the inner ear is comprised of the cochlea and vestibular system, which develop from the otic vesicle. This process is regulated via inductive interactions from surrounding tissues. Tbx1, the gene responsible for velo-cardio-facial syndrome/DiGeorge syndrome in humans, is required for ear development in mice. Tbx1 is expressed in the otic epithelium and adjacent periotic mesenchyme (POM), and both of these domains are required for inner ear formation. To study the function of Tbx1 in the POM, we have conditionally inactivated Tbx1 in the mesoderm while keeping expression in the otic vesicle intact.
resultsConditional mutants (TCre-KO) displayed malformed inner ears, including a hypoplastic otic vesicle and a severely shortened cochlear duct, indicating that Tbx1 expression in the POM is necessary for proper inner ear formation. Expression of the mesenchyme marker Brn4 was also lost in the TCre-KO. Brn4-;Tbx1+/-embryos displayed defects in growth of the distal cochlea. To identify a potential signal from the POM to the otic epithelium, expression of retinoic acid (RA) catabolizing genes was examined in both mutants. Cyp26a1 expression was altered in the TCre-KO, while Cyp26c1 showed reduced expression in both TCre-KO and Brn4-;Tbx1+/- embryos.
conclusionThese results indicate that Tbx1 expression in the POM regulates cochlear outgrowth potentially via control of local retinoic acid activity.
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