title: RFX7 is required for the formation of cilia in the neural tube.
abstract: Regulatory Factor X (RFX) transcription factors are important for development and are likely involved in the pathogenesis of serious human diseases including ciliopathies. While seven RFX genes have been identified in vertebrates and several RFX transcription factors have been reported to be regulators of ciliogenesis, the role of RFX7 in development including ciliogenesis is not known. Here we show that RFX7 in Xenopus laevis is expressed in the neural tube, eye, otic vesicles, and somites. Knockdown of RFX7 in Xenopus embryos resulted in a defect of ciliogenesis in the neural tube and failure of neural tube closure. RFX7 controlled the formation of cilia by regulating the expression of RFX4 gene, which has been reported to be required for ciliogenesis in the neural tube. Moreover, ectopic expression of Foxj1, which is a master regulator of motile cilia formation, suppressed the expression of RFX4 but not RFX7. Taken together, RFX7 plays an important role in the process of neural tube closure at the top of the molecular cascade which controls ciliogenesis in the neural tube.
authors: Department of Biomedical Sciences, Florida State University College of Medicine, Tallahassee, FL 32306, USA.
authors: Zarko
authors: Z
authors: Manojlovic
authors: Department of Biomedical Sciences, Florida State University College of Medicine, Tallahassee, FL 32306, USA.
authors: Ryan
authors: R
authors: Earwood
authors: Department of Biomedical Sciences, Florida State University College of Medicine, Tallahassee, FL 32306, USA.
authors: Akiko
authors: A
authors: Kato
authors: Department of Biomedical Sciences, Florida State University College of Medicine, Tallahassee, FL 32306, USA. Electronic address: branko.stefanovic@med.fsu.edu.
authors: Branko
authors: B
authors: Stefanovic
authors: Department of Biomedical Sciences, Florida State University College of Medicine, Tallahassee, FL 32306, USA. Electronic address: yoichi.kato@med.fsu.edu.
authors: Yoichi
authors: Y
authors: Kato
mesh_terms: D000818:Animals; D001483:Base Sequence; D002923:Cilia / Q000235:genetics*; D004268:DNA-Binding Proteins / Q000235:genetics*; D051858:Forkhead Transcription Factors / Q000235:genetics; D018507:Gene Expression Regulation, Developmental / Q000235:genetics; D008969:Molecular Sequence Data; D009024:Morphogenesis / Q000235:genetics; D054259:Neural Tube / Q000196:embryology*; D000071701:Regulatory Factor X Transcription Factors; D014157:Transcription Factors / Q000235:genetics*; D029867:Xenopus Proteins / Q000235:genetics; D014982:Xenopus laevis
chemical_list: D004268:DNA-Binding Proteins; C490956:FOXJ1 protein, Xenopus; D051858:Forkhead Transcription Factors; D000071701:Regulatory Factor X Transcription Factors; D014157:Transcription Factors; D029867:Xenopus Proteins
keywords: Cilia; Neural tube closure; RFX; Xenopus laevis