Notch/Her12 signalling modulates, motile/immotile cilia ratio downstream of Foxj1a in zebrafish left-right organizer

Tavares, Bárbara; Jacinto, Raquel; Sampaio, Pedro; Pestana, Sara; Pinto, Andreia; Vaz, Andreia; Roxo-Rosa, Mónica; Gardner, Rui; Lopes, Telma; Schilling, Britta; Henry, Ian; Saúde, Leonor; Lopes, Susana S.

doi:10.7554/elife.25165

Cited by 29 publications

(52 citation statements)

References 57 publications

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“…Zebrafish studies on the speed of flow per KV regions have shown that flow has a stereotyped pattern that is biologically relevant for LR establishment [2]. When we stopped motility of cilia everywhere except in the right half of the KV, we could demonstrate that this led to larva with situs inversus [9]. Although this seems to advocate for a mechanosensation process, it is still possible that some chemical component exists.…”

Section: The Kv As a Model Organ To Study Pcdmentioning

confidence: 81%

“…The Vermot lab by pioneering the studies on KV flow [40] and recently by studying cilia tilt along the 3D KV [12] and its resultant theoretical flow forces. Contradictory interpretations emerged from our lab [9] and Vermot lab [12] concerning the number of immotile cilia in the KV and the consequent ability for the animal to sense flow in a mechanosensory way. This subject needs further experiments to be solved as our labs used different imaging protocols to determine the number of immotile cilia.…”

Section: The Kv As a Model Organ To Study Pcdmentioning

confidence: 95%

“…CFTR is a chloride channel which absence or dysfunction causes cystic fibrosis [6][7][8]. As the KV enlarges, the solitary cilium from each KV cell starts to beat, and by ten somite stage (ss), there are on average 80% motile cilia and 20% immotile cilia in a total of 60 cells [9]. Despite it presents different shapes and sizes, the left-right organizer (LRO) has a conserved function among vertebrates, which is to break initial symmetry of embryonic body plans.…”

Section: The Kupffer's Vesiclementioning

confidence: 99%

“…As detailed below there are still many gaps in the developmental process of left-right (LR) establishment that need to be tested. Due to its genetic amenability and optical transparency, the zebrafish has gained impact in the LR field as one of the best models for testing early LR establishment questions [2,[9][10][11][12][13][14][15]. So far, it is the only animal system where we can manipulate the LRO without damaging or sacrificing the animal, thus letting it develop until organ localization can be visualized [2].…”

Section: The Kupffer's Vesiclementioning

confidence: 99%

“…As already mentioned, this fluid-filled vesicle organ is lined by monociliated cells [1,2,9]. The KV cilia are motile and generate a fluid flow that is essential for the organ function [2,9].…”

Section: The Zebrafish Pkd Genesmentioning

confidence: 99%

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The Zebrafish Kupffer’s Vesicle: A Special Organ in a Model Organism to Study Human Diseases

Roxo-Rosa¹,

Lopes²

2020

Zebrafish in Biomedical Research

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The Kupffer's vesicle (KV) is a small, ciliated organ transiently present during embryogenesis of the zebrafish and other teleosts. The KV is required to the establishment of visceral laterality, such as the heart on the left side, being also known by the name left-right organizer (LRO). The LRO is found in other vertebrates, including mice, rabbits, frogs and human embryos. Among these, the KV became an excellent model organ to investigate the early left-right events during development and in disease. Many ciliary molecular players associated to the human disease primary ciliary dyskinesia have been tested in the zebrafish looking at KV cilia and its downstream effects on flow and left-right markers. Additionally, given its morphology and molecular features, we proposed the KV as a model organ to study the molecular mechanisms of the renal cyst inflation that occurs in the autosomal dominant polycystic kidney disease. Although having no connection to the kidney, the KV mimics a renal cyst because it is a fluid-filled vesicle, lined by monociliated epithelial cells that express polycystin-2, which knockdown leads to the organ luminal enlargement through changes in ion/water epithelial transport. Here, we explore the usefulness of the zebrafish KV to model these diseases. Keywords: Kupffer's vesicle (KV), left-right (LR), left-right organizer (LRO), primary ciliary dyskinesia (PCD), autosomal dominant polycystic kidney disease (ADPKD) Zebrafish in Biomedical Research

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Section: The Kv As a Model Organ To Study Pcdmentioning

confidence: 81%

Section: The Kv As a Model Organ To Study Pcdmentioning

confidence: 95%

Section: The Kupffer's Vesiclementioning

confidence: 99%

Section: The Kupffer's Vesiclementioning

confidence: 99%

Section: The Zebrafish Pkd Genesmentioning

confidence: 99%

See 3 more Smart Citations

The Zebrafish Kupffer’s Vesicle: A Special Organ in a Model Organism to Study Human Diseases

Roxo-Rosa¹,

Lopes²

2020

Zebrafish in Biomedical Research

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Structures and functions of cilia during vertebrate embryo development

Amack

2022

Molecular Reproduction Devel

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Cilia are hair‐like structures that project from the surface of cells. In vertebrates, most cells have an immotile primary cilium that mediates cell signaling, and some specialized cells assemble one or multiple cilia that are motile and beat synchronously to move fluids in one direction. Gene mutations that alter cilia structure or function cause a broad spectrum of disorders termed ciliopathies that impact virtually every system in the body. A wide range of birth defects associated with ciliopathies underscores critical functions for cilia during embryonic development. In many cases, the mechanisms underlying cilia functions during development and disease remain poorly understood. This review describes different types of cilia in vertebrate embryos and discusses recent research results from diverse model systems that provide novel insights into how cilia form and function during embryo development. The work discussed here not only expands our understanding of in vivo cilia biology, but also opens new questions about cilia and their roles in establishing healthy embryos.

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Wall stress enhanced exocytosis of extracellular vesicles as a possible mechanism of left-right symmetry-breaking in vertebrate development

Solowiej-Wedderburn

Smith

Lopes

et al. 2019

Journal of Theoretical Biology

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Where a licence is displayed above, please note the terms and conditions of the licence govern your use of this document. When citing, please reference the published version. Take down policy While the University of Birmingham exercises care and attention in making items available there are rare occasions when an item has been uploaded in error or has been deemed to be commercially or otherwise sensitive.

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Notch/Her12 signalling modulates, motile/immotile cilia ratio downstream of Foxj1a in zebrafish left-right organizer

Cited by 29 publications

References 57 publications

The Zebrafish Kupffer’s Vesicle: A Special Organ in a Model Organism to Study Human Diseases

The Zebrafish Kupffer’s Vesicle: A Special Organ in a Model Organism to Study Human Diseases

Structures and functions of cilia during vertebrate embryo development

Wall stress enhanced exocytosis of extracellular vesicles as a possible mechanism of left-right symmetry-breaking in vertebrate development

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