PUBLICATION

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

Authors: Barrodia, P., Patra, C., Swain, R.K.
ID: ZDB-PUB-181024-6
Date: 2018
Source: Cell & Bioscience 8: 53 (Journal)
Registered Authors
Keywords: Efhc2, Multi-ciliated cells, Pronephros, Retinoic acid, Zebrafish
MeSH Terms: none
PubMed: 30349665 Full text @ Cell Biosci.

Abstract

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.

Genes / Markers

Figures

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Expression

Gene	Fish	Conditions	Stage	Anatomy	Assay	Figure
cimap1b	WT	control	Long-pec	pronephros multi-ciliated epithelial cell	ISH	Fig. 7
cimap1b	WT	chemical treatment by environment: retinoic acid	Long-pec	pronephros multi-ciliated epithelial cell	ISH	Fig. 7
cimap1b	WT	chemical treatment by environment: citral	Long-pec	pronephros multi-ciliated epithelial cell	ISH	Fig. 7
cimap1b	WT	chemical treatment by environment: 3-allyl-2-[2-(diethylamino)ethoxy]benzaldehyde	Long-pec	pronephros multi-ciliated epithelial cell	ISH	Fig. 7
cimap1b	WT + MO1-efhc2	chemical treatment by environment: retinoic acid	Long-pec	pronephros multi-ciliated epithelial cell	ISH	Fig. 7
cimap1b	WT + MO1-efhc2	chemical treatment by environment: citral	Long-pec	pronephros multi-ciliated epithelial cell	ISH	Fig. 7
cimap1b	WT + MO1-efhc2	control	Long-pec	pronephros multi-ciliated epithelial cell	ISH	Fig. 7
cimap1b	WT + MO1-efhc2	chemical treatment by environment: 3-allyl-2-[2-(diethylamino)ethoxy]benzaldehyde	Long-pec	pronephros multi-ciliated epithelial cell	ISH	Fig. 7
efhc2	WT	standard conditions	Prim-5	epiphysis	ISH	Fig. 1
efhc2	WT	standard conditions	Prim-25	epiphysis	ISH	Fig. 1

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Phenotype

Fish	Conditions	Stage	Phenotype	Figure
WT	chemical treatment by environment: citral	Long-pec	corpuscles of Stannius stc1 expression mislocalised, abnormal	Fig. 6
WT	chemical treatment by environment: citral	Long-pec	pronephric distal early tubule slc12a1 expression mislocalised, abnormal	Fig. 6
WT	chemical treatment by environment: citral	Long-pec	pronephric distal late tubule slc12a3 expression increased distribution, abnormal	Fig. 6
WT	chemical treatment by environment: citral	Long-pec	pronephric distal late tubule slc12a3 expression mislocalised, abnormal	Fig. 6
WT	chemical treatment by environment: citral	Long-pec	pronephros multi-ciliated epithelial cell cimap1b expression decreased distribution, abnormal	Fig. 7
WT	chemical treatment by environment: 3-allyl-2-[2-(diethylamino)ethoxy]benzaldehyde	Long-pec	corpuscles of Stannius stc1 expression mislocalised, abnormal	Fig. 5
WT	chemical treatment by environment: 3-allyl-2-[2-(diethylamino)ethoxy]benzaldehyde	Long-pec	corpuscles of Stannius stc1 expression spatial pattern, abnormal	Fig. 5
WT	chemical treatment by environment: 3-allyl-2-[2-(diethylamino)ethoxy]benzaldehyde	Long-pec	corpuscles of Stannius mislocalised proximally, abnormal	Fig. 5
WT	chemical treatment by environment: 3-allyl-2-[2-(diethylamino)ethoxy]benzaldehyde	Long-pec	pronephric distal early tubule slc12a1 expression mislocalised, abnormal	Fig. 5
WT	chemical treatment by environment: 3-allyl-2-[2-(diethylamino)ethoxy]benzaldehyde	Long-pec	pronephric distal early tubule slc12a1 expression spatial pattern, abnormal	Fig. 5

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Mutations / Transgenics

Human Disease / Model

Sequence Targeting Reagents

Target	Reagent	Reagent Type
efhc2	MO1-efhc2	MRPHLNO
efhc2	MO2-efhc2	MRPHLNO

Fish

Fish
WT
WT + MO1-efhc2
WT + MO2-efhc2

Antibodies

Orthology

Engineered Foreign Genes

Mapping

Barrodia et al., 2018 ZDB-PUB-181024-6 PMID:30349665

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

Barrodia et al., 2018

ZDB-PUB-181024-6
PMID:30349665