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Neural crest-specific deletion of Rbfox2 in mice leads to craniofacial abnormalities including cleft palate

التفاصيل البيبلوغرافية
العنوان: Neural crest-specific deletion of Rbfox2 in mice leads to craniofacial abnormalities including cleft palate
المؤلفون: Dasan Mary Cibi, Masum M Mia, Shamini Guna Shekeran, Lim Sze Yun, Reddemma Sandireddy, Priyanka Gupta, Monalisa Hota, Lei Sun, Sujoy Ghosh, Manvendra K Singh
المصدر: eLife, Vol 8 (2019)
بيانات النشر: eLife Sciences Publications Ltd, 2019.
سنة النشر: 2019
المجموعة: LCC:Medicine
LCC:Science
LCC:Biology (General)
مصطلحات موضوعية: alternative splicing, neural crest, Rbfox2, cleft palate, TGF-β signaling, Tak1, Medicine, Science, Biology (General), QH301-705.5
الوصف: Alternative splicing (AS) creates proteomic diversity from a limited size genome by generating numerous transcripts from a single protein-coding gene. Tissue-specific regulators of AS are essential components of the gene regulatory network, required for normal cellular function, tissue patterning, and embryonic development. However, their cell-autonomous function in neural crest development has not been explored. Here, we demonstrate that splicing factor Rbfox2 is expressed in the neural crest cells (NCCs), and deletion of Rbfox2 in NCCs leads to cleft palate and defects in craniofacial bone development. RNA-Seq analysis revealed that Rbfox2 regulates splicing and expression of numerous genes essential for neural crest/craniofacial development. We demonstrate that Rbfox2-TGF-β-Tak1 signaling axis is deregulated by Rbfox2 deletion. Furthermore, restoration of TGF-β signaling by Tak1 overexpression can rescue the proliferation defect seen in Rbfox2 mutants. We also identified a positive feedback loop in which TGF-β signaling promotes expression of Rbfox2 in NCCs.
نوع الوثيقة: article
وصف الملف: electronic resource
اللغة: English
تدمد: 2050-084X
Relation: https://elifesciences.org/articles/45418; https://doaj.org/toc/2050-084X
DOI: 10.7554/eLife.45418
URL الوصول: https://doaj.org/article/affddb8f910c4c8e8b0e4a595711c167
رقم الأكسشن: edsdoj.ffddb8f910c4c8e8b0e4a595711c167
قاعدة البيانات: Directory of Open Access Journals
الوصف
تدمد:2050084X
DOI:10.7554/eLife.45418