PUBLICATION
Leucine-rich repeat containing 8A contributes to the expansion of brain ventricles in zebrafish embryos
- Authors
- Tseng, Y.T., Ko, C.L., Chang, C.T., Lee, Y.H., Huang Fu, W.C., Liu, I.H.
- ID
- ZDB-PUB-200118-1
- Date
- 2020
- Source
- Biology Open 9(1): (Journal)
- Registered Authors
- Lee, Yen-Hua, Liu, I-Hsuan, Tseng, Yen-Tzu
- Keywords
- Lrrc8A, Organic osmoregulation, Taurine, Volume-regulated anion channel (VRAC), Volume-sensitive organic osmolyte/anion channel (VSOAC)
- MeSH Terms
-
- Animals
- Astrocytes/cytology
- Astrocytes/metabolism
- Brain*/cytology
- Brain*/embryology
- Embryo, Nonmammalian*/cytology
- Embryo, Nonmammalian*/embryology
- Gene Expression Regulation, Developmental*
- Ion Channels*/biosynthesis
- Ion Channels*/genetics
- Osmoregulation/physiology*
- Zebrafish*/embryology
- Zebrafish*/genetics
- Zebrafish Proteins*/biosynthesis
- Zebrafish Proteins*/genetics
- PubMed
- 31941702 Full text @ Biol. Open
Citation
Tseng, Y.T., Ko, C.L., Chang, C.T., Lee, Y.H., Huang Fu, W.C., Liu, I.H. (2020) Leucine-rich repeat containing 8A contributes to the expansion of brain ventricles in zebrafish embryos. Biology Open. 9(1):.
Abstract
The sodium osmotic gradient is necessary for the initiation of brain ventricle inflation, but a previous study predicted that organic and inorganic osmolytes play equivalently important roles in osmotic homeostasis in astrocytes. To test whether organic osmoregulation also plays a role in brain ventricle inflation, the core component for volume-regulated anion and organic osmolyte channel, lrrc8a, was investigated in zebrafish model. RT-PCR and whole-mount in situ hybridization indicated that both genes were ubiquitously expressed through 12 hpf, and around the ventricular layer of neural tubes and the cardiogenic region at 24 hpf. Knocking down either one lrrc8a paralog with morpholino oligos resulted in abnormalities in circulation at 32 hpf. Morpholino oligos or CRISPR interference against either paralog led to smaller brain ventricles at 24 hpf. Either lrrc8aa or lrrc8ab mRNA rescued the phenotypic penetrance in both lrrc8aa and lrrc8ab morphants. Supplementation of taurine in the E3 medium and overexpression csad mRNA also rescued lrrc8aa and lrrc8ab morphants. Our results indicated that the two zebrafish lrrc8a paralogs are maternal message genes and were ubiquitously expressed in early embryos. The two genes play redundant roles in the expansion of brain ventricles and the circulatory system and taurine contribute to the brain ventricle expansion via volume-regulated anion and organic osmolyte channel.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping