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EF-hand domain containing 2 (Efhc2) is crucial for distal segmentation of pronephros in zebrafish

BACKGROUND: The blood filtering organ in zebrafish embryos is the pronephros, which consists of two functional nephrons. Segmentation of a nephron into different domains is essential for its function and is well conserved among vertebrates. Zebrafish has been extensively used as a model to understan...

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Autores principales: Barrodia, Praveen, Patra, Chinmoy, Swain, Rajeeb K.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6192171/
https://www.ncbi.nlm.nih.gov/pubmed/30349665
http://dx.doi.org/10.1186/s13578-018-0253-z
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author Barrodia, Praveen
Patra, Chinmoy
Swain, Rajeeb K.
author_facet Barrodia, Praveen
Patra, Chinmoy
Swain, Rajeeb K.
author_sort Barrodia, Praveen
collection PubMed
description BACKGROUND: The blood filtering organ in zebrafish embryos is the pronephros, which consists of two functional nephrons. Segmentation of a nephron into different domains is essential for its function and is well conserved among vertebrates. Zebrafish has been extensively used as a model to understand nephron segmentation during development. Here, we have identified EF-hand domain containing 2 (Efhc2) as a novel component of genetic programme regulating nephron segmentation in zebrafish. Human EFHC2 is a protein with one predicted calcium-binding EF-hand motif and three DM10 domains, whose function is unknown. EFHC2 has been implicated in several brain-related genetic diseases like Turner syndrome and juvenile myoclonic epilepsy. However, there is limited information on its normal physiological function. RESULTS: efhc2 mRNA is primarily expressed in the pronephros of zebrafish embryos. Other sites of expression include olfactory placode, notochord, otic vesicle, epiphysis and neuromast cells. Morpholino antisense oligonucleotide-mediated knock-down of Efhc2 resulted in defects in pronephros development and function in zebrafish embryos. Efhc2 knock-down leads to expansion of distal early segment of pronephros, whereas, the corpuscle of stannius and distal late segments were reduced. The number of multi-ciliated cells (MCC) that are present in a salt-and-pepper fashion throughout the middle of each nephron and vital for fluid flow were also reduced. It is known that retinoic acid (RA) signaling regulates pronephros segmentation in vertebrates and we show that Efhc2 function is crucial for nephron segmentation in zebrafish. Our data suggests that RA and Efhc2 function independent of each other in pronephros segmentation. However, Efhc2 and RA synergistically regulate MCC development. CONCLUSION: In this study, we have identified Efhc2 as a regulator of segmentation of the distal part of nephron and pronephros function during zebrafish development. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (10.1186/s13578-018-0253-z) contains supplementary material, which is available to authorized users.
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spelling pubmed-61921712018-10-22 EF-hand domain containing 2 (Efhc2) is crucial for distal segmentation of pronephros in zebrafish Barrodia, Praveen Patra, Chinmoy Swain, Rajeeb K. Cell Biosci Research BACKGROUND: The blood filtering organ in zebrafish embryos is the pronephros, which consists of two functional nephrons. Segmentation of a nephron into different domains is essential for its function and is well conserved among vertebrates. Zebrafish has been extensively used as a model to understand nephron segmentation during development. Here, we have identified EF-hand domain containing 2 (Efhc2) as a novel component of genetic programme regulating nephron segmentation in zebrafish. Human EFHC2 is a protein with one predicted calcium-binding EF-hand motif and three DM10 domains, whose function is unknown. EFHC2 has been implicated in several brain-related genetic diseases like Turner syndrome and juvenile myoclonic epilepsy. However, there is limited information on its normal physiological function. RESULTS: efhc2 mRNA is primarily expressed in the pronephros of zebrafish embryos. Other sites of expression include olfactory placode, notochord, otic vesicle, epiphysis and neuromast cells. Morpholino antisense oligonucleotide-mediated knock-down of Efhc2 resulted in defects in pronephros development and function in zebrafish embryos. Efhc2 knock-down leads to expansion of distal early segment of pronephros, whereas, the corpuscle of stannius and distal late segments were reduced. The number of multi-ciliated cells (MCC) that are present in a salt-and-pepper fashion throughout the middle of each nephron and vital for fluid flow were also reduced. It is known that retinoic acid (RA) signaling regulates pronephros segmentation in vertebrates and we show that Efhc2 function is crucial for nephron segmentation in zebrafish. Our data suggests that RA and Efhc2 function independent of each other in pronephros segmentation. However, Efhc2 and RA synergistically regulate MCC development. CONCLUSION: In this study, we have identified Efhc2 as a regulator of segmentation of the distal part of nephron and pronephros function during zebrafish development. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (10.1186/s13578-018-0253-z) contains supplementary material, which is available to authorized users. BioMed Central 2018-10-16 /pmc/articles/PMC6192171/ /pubmed/30349665 http://dx.doi.org/10.1186/s13578-018-0253-z Text en © The Author(s) 2018 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Research
Barrodia, Praveen
Patra, Chinmoy
Swain, Rajeeb K.
EF-hand domain containing 2 (Efhc2) is crucial for distal segmentation of pronephros in zebrafish
title EF-hand domain containing 2 (Efhc2) is crucial for distal segmentation of pronephros in zebrafish
title_full EF-hand domain containing 2 (Efhc2) is crucial for distal segmentation of pronephros in zebrafish
title_fullStr EF-hand domain containing 2 (Efhc2) is crucial for distal segmentation of pronephros in zebrafish
title_full_unstemmed EF-hand domain containing 2 (Efhc2) is crucial for distal segmentation of pronephros in zebrafish
title_short EF-hand domain containing 2 (Efhc2) is crucial for distal segmentation of pronephros in zebrafish
title_sort ef-hand domain containing 2 (efhc2) is crucial for distal segmentation of pronephros in zebrafish
topic Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6192171/
https://www.ncbi.nlm.nih.gov/pubmed/30349665
http://dx.doi.org/10.1186/s13578-018-0253-z
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