Zebrafish Rfx4 controls dorsal and ventral midline formation in the neural tube

Sedykh, Irina; Keller, Abigail N.; Yoon, Baul; Roberson, Laura; Moskvin, Oleg V.; Grinblat, Yevgenya

doi:10.1002/dvdy.24613

Cited by 22 publications

(21 citation statements)

References 67 publications

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“…We used shha as probe to target mRNA in the floor plate. Shha is expressed in the cranial primordium and in the spinal cord floor plate, adjacent to notochord [17]. We also show that the floor plate marker is strongly expressed in these structures at 20 hours post fertilization (hpf) (Fig 7A-7C) and 1 dpf (Fig 7D-7F).…”

Section: Plos Onesupporting

confidence: 55%

All-age whole mount in situ hybridization to reveal larval and juvenile expression patterns in zebrafish

et al. 2020

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The zebrafish Danio rerio is a valuable and common model for scientists in the fields of genetics and developmental biology. Since zebrafish are also amenable to genetic manipulation, modelling of human diseases or behavioral experiments have moved into the focus of zebrafish research. Consequently, gene expression data beyond embryonic and larval stages become more important, yet there is a dramatic knowledge gap of gene expression beyond day four of development. Like in other model organisms, the visualization of spatial and temporal gene expression by whole mount in situ hybridization (ISH) becomes increasingly difficult when zebrafish embryos develop further and hence the growing tissues become dense and less permeable. Here we introduce a modified method for whole mount ISH, which overcomes these penetration and detection problem. The method is an all in one solution that enables the detection and visualization of gene expression patterns up to the late larval stage in a 3D manner without the need for tissue sectioning and offers a valuable extension for whole mount ISH by immunohistochemistry in the zebrafish field.

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Section: Plos Onesupporting

confidence: 55%

All-age whole mount in situ hybridization to reveal larval and juvenile expression patterns in zebrafish

et al. 2020

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“…Using animal models such as the mouse, chick, Xenopus, and zebrafish, researchers have successfully constructed many NTD models to study the underlying mechanisms of failed NTC. [37][38][39][40] Many transgenic mouse models have been created to study NTDs, which has been reviewed in many recently published papers. 15,25,[41][42][43] However, these animal models cannot fully represent human NTDs, as not only are their genomes different from human's but also the formation of the neural tube are not completely identical among different model species.…”

Section: Animal Models For Ntdsmentioning

confidence: 99%

Organoids as a new model system to study neural tube defects

Peng

Finnell

et al. 2021

The FASEB Journal

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The neural tube is the first critically important structure that develops in the embryo. It serves as the primordium of the central nervous system; therefore, the proper formation of the neural tube is essential to the developing organism. Neural tube defects (NTDs) are severe congenital defects caused by failed neural tube closure during early embryogenesis. The pathogenesis of NTDs is complicated and still not fully understood even after decades of research. While it is an ethically impossible proposition to investigate the in vivo formation process of the neural tube in human embryos, a newly developed technology involving the creation of neural tube organoids serves as an excellent model system with which to study human neural tube formation and the occurrence of NTDs. Herein we reviewed the recent literature on the process of neural tube formation, the progress of NTDs investigations, and particularly the exciting potential to use neural tube organoids to model the cellular and molecular mechanisms underlying the etiology of NTDs.

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“…In addition to their conserved roles in ciliogenesis, RFX proteins are involved in other processes ranging from the control of genome integrity and the cell cycle in yeast and fungi ( 34 , 35 ), to immune functions ( 36 , 37 ), and tissue and organ differentiation in the mouse or zebrafish ( 33 , 38–40 ). Several human diseases result from mutations in Rfx genes ( 37 , 38 , 41 ).…”

Section: Introductionmentioning

confidence: 99%

Interplay of RFX transcription factors 1, 2 and 3 in motile ciliogenesis

Lemeille

Paschaki

Baas

et al. 2020

Nucleic Acids Research

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Cilia assembly is under strict transcriptional control during animal development. In vertebrates, a hierarchy of transcription factors (TFs) are involved in controlling the specification, differentiation and function of multiciliated epithelia. RFX TFs play key functions in the control of ciliogenesis in animals. Whereas only one RFX factor regulates ciliogenesis in C. elegans, several distinct RFX factors have been implicated in this process in vertebrates. However, a clear understanding of the specific and redundant functions of different RFX factors in ciliated cells remains lacking. Using RNA-seq and ChIP-seq approaches we identified genes regulated directly and indirectly by RFX1, RFX2 and RFX3 in mouse ependymal cells. We show that these three TFs have both redundant and specific functions in ependymal cells. Whereas RFX1, RFX2 and RFX3 occupy many shared genomic loci, only RFX2 and RFX3 play a prominent and redundant function in the control of motile ciliogenesis in mice. Our results provide a valuable list of candidate ciliary genes. They also reveal stunning differences between compensatory processes operating in vivo and ex vivo.

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Zebrafish Rfx4 controls dorsal and ventral midline formation in the neural tube

Cited by 22 publications

References 67 publications

All-age whole mount in situ hybridization to reveal larval and juvenile expression patterns in zebrafish

All-age whole mount in situ hybridization to reveal larval and juvenile expression patterns in zebrafish

Organoids as a new model system to study neural tube defects

Interplay of RFX transcription factors 1, 2 and 3 in motile ciliogenesis

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