PUBLICATION

Cystein-serine-rich nuclear protein 1, Axud1/Csrnp1, is essential for cephalic neural progenitor proliferation and survival in zebrafish

Authors

Feijoo, C.G., Sarrazin, A.F., Allende, M.L., and Glavic, A.

ID

ZDB-PUB-090629-18

Date

2009

Source

Developmental Dynamics : an official publication of the American Association of Anatomists 238(8): 2034-2043 (Journal)

Registered Authors

Allende, Miguel L., Feijoo, Carmen G., Sarrazin, Andrés

Keywords

axud1, brain growth, neural progenitors, proliferation, cell survival

MeSH Terms

Homeodomain Proteins/genetics
Homeodomain Proteins/physiology
Mice
Zebrafish Proteins/genetics
Zebrafish Proteins/physiology*
Amino Acid Sequence
Eye Proteins/genetics
Eye Proteins/physiology
Embryonic Stem Cells/cytology
Sequence Homology, Amino Acid
Neurons/cytology
Gene Targeting
Cell Survival/genetics
Cell Survival/physiology
Zebrafish/embryology*
Zebrafish/genetics
Zebrafish/physiology*
Hedgehog Proteins/genetics
Hedgehog Proteins/physiology
Gene Expression Regulation, Developmental
Humans
Nuclear Proteins/genetics
Nuclear Proteins/physiology*
Body Patterning/genetics
Body Patterning/physiology
Phylogeny
Brain/cytology
Brain/embryology*
Cell Proliferation
Animals
Molecular Sequence Data
Nerve Tissue Proteins/genetics
Nerve Tissue Proteins/physiology
Base Sequence
DNA, Complementary/genetics

(all 35)

PubMed

19544579 Full text @ Dev. Dyn.

Abstract

The CSRNP (cystein-serine-rich nuclear protein) family has been conserved from Drosophila to human. Although knockout mice for each of the mammalian proteins have been generated, their function during vertebrate development has remained elusive. As an alternative to obtain insights on CSRNP's role in development, we have analysed the expression pattern and function of one member of this family, axud1, during zebrafish development. Our expression analysis indicates that axud1 is expressed from cleavage to larval stages in a dynamic pattern, becoming restricted after gastrulation to anterior regions of the developing neuraxis and later on concentrated predominantly in proliferating domains of the brain. Knockdown analysis using antisense morpholinos shows that reducing Axud1 levels impairs neural progenitor cell proliferation and survival, revealing an essential function of this gene for the growth of cephalic derivatives. The brain growth phenotypes elicited by decreasing Axud1 expression are specific and independent of anterior-posterior patterning events, initial establishment of neural progenitors, or neural differentiation occurring in this tissue. However, Axud1 is necessary for six3.1 expression and is positively regulated by sonic hedgehog. Phylogenetic examination shows that axud1 is likely to be the ortholog of the only member of this family present in Drosophila, as well as to the previously described mouse CSRNP1 and to human AXUD1 (Axin upregulated-1). Thus, we provide evidence as to the role of axud1 in brain growth in vertebrates.

Genes / Markers

Figures

Show all Figures

Expression

Gene	Fish	Conditions	Stage	Qualifier	Anatomy	Assay	Figure
csrnp1b	smo^b577/b577	standard conditions	Prim-5	Not Detected	forebrain	ISH	Fig. 6
csrnp1b	smo^b577/b577	standard conditions	Prim-5	Not Detected	hindbrain	ISH	Fig. 6
csrnp1b	smo^b577/b577	standard conditions	Prim-5	Not Detected	midbrain hindbrain boundary	ISH	Fig. 6
csrnp1b	smo^b577/b577	standard conditions	Prim-5	Not Detected	optic tectum	ISH	Fig. 6
csrnp1b	WT	standard conditions	Bud		axial chorda mesoderm	ISH	Fig. 2
csrnp1b	WT	standard conditions	14-19 somites		diencephalon	ISH	Fig. 2
csrnp1b	WT	standard conditions	14-19 somites		epiphysis	ISH	Fig. 2
csrnp1b	WT	standard conditions	26+ somites		forebrain	ISH	Fig. 2
csrnp1b	WT	standard conditions	Prim-5		forebrain	ISH	Fig. 6
csrnp1b	WT	standard conditions	20-25 somites		forebrain	ISH	Fig. 2

1 - 10 of 58

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Phenotype

Fish	Conditions	Stage	Phenotype	Figure
WT + MO1-csrnp1b	standard conditions	Bud	cell proliferation in midbrain decreased rate, abnormal	Fig. 4
WT + MO1-csrnp1b	standard conditions	Bud	neuron apoptotic process increased rate, abnormal	Fig. 4
WT + MO1-csrnp1b	standard conditions	Prim-5	cell proliferation in midbrain decreased rate, abnormal	Fig. 6
WT + MO1-csrnp1b	standard conditions	Prim-5	eye decreased size, abnormal	Fig. 3
WT + MO1-csrnp1b	standard conditions	Prim-5	forebrain physical object quality, abnormal	Fig. 6
WT + MO1-csrnp1b	standard conditions	Prim-5	head decreased size, abnormal	Fig. 3
WT + MO1-csrnp1b	standard conditions	Prim-5	midbrain hindbrain boundary malformed, abnormal	Fig. 3
WT + MO1-csrnp1b	standard conditions	Prim-5	midbrain hindbrain boundary physical object quality, abnormal	Fig. 6
WT + MO1-csrnp1b	standard conditions	Prim-5	neuron apoptotic process increased rate, abnormal	Fig. 6
WT + MO1-csrnp1b	standard conditions	Prim-5	otic vesicle decreased size, abnormal	Fig. 3

1 - 10 of 16

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Mutations / Transgenics

Allele	Construct	Type	Affected Genomic Region
b577		Point Mutation	smo

Human Disease / Model

Sequence Targeting Reagents

Target	Reagent	Reagent Type
csrnp1b	MO1-csrnp1b	MRPHLNO
csrnp1b	MO2-csrnp1b	MRPHLNO

Fish

Fish
smo^b577/b577
WT
WT + MO1-csrnp1b
WT + MO2-csrnp1b

Antibodies

Name	Type	Antigen Genes	Isotypes	Host Organism
Ab6-h3	polyclonal			Rabbit

Orthology

Gene	Orthology
csrnp1b

Engineered Foreign Genes

Mapping

Feijoo et al., 2009 ZDB-PUB-090629-18 PMID:19544579

Cystein-serine-rich nuclear protein 1, Axud1/Csrnp1, is essential for cephalic neural progenitor proliferation and survival in zebrafish

Feijoo et al., 2009

ZDB-PUB-090629-18
PMID:19544579