PUBLICATION

Zebrafish sip1a and sip1b are essential for normal axial and neural patterning

Authors

Delalande, J.M., Guyote, M.E., Smith, C.M., and Shepherd, I.T.

ID

ZDB-PUB-080327-2

Date

2008

Source

Developmental Dynamics : an official publication of the American Association of Anatomists 237(4): 1060-1069 (Journal)

Registered Authors

Shepherd, Iain T.

Keywords

zebrafish, enteric nervous system, SIP1, Mowat-Wilson syndrome, neural crest

MeSH Terms

Animals
Stem Cells/physiology
Body Patterning*
Base Sequence
Zebrafish Proteins/genetics
Zebrafish Proteins/metabolism*
Molecular Sequence Data
Gene Expression Regulation, Developmental
Protein Isoforms/genetics
Protein Isoforms/metabolism*
Humans
Oligonucleotides, Antisense/genetics
Oligonucleotides, Antisense/metabolism
Morphogenesis
Phenotype
In Situ Hybridization
Zebrafish*/anatomy & histology
Zebrafish*/embryology
Zebrafish*/metabolism
Neural Crest/cytology
Carrier Proteins/genetics
Carrier Proteins/metabolism*

(all 22)

PubMed

18351671 Full text @ Dev. Dyn.

Abstract

Smad-interacting protein-1 (SIP1) has been implicated in the development of Mowat-Wilson syndrome whose patients exhibit Hirschsprung disease, an aganglionosis of the large intestine, as well as other phenotypes. We have identified and cloned two sip1 orthologues in zebrafish. Both sip1 orthologues are expressed maternally and have dynamic zygotic expression patterns that are temporally and spatially distinct. We have investigated the function of both orthologues using translation and splice-blocking morpholino antisense oligonucleotides. Knockdown of the orthologues causes axial and neural patterning defects consistent with the previously described function of SIP1 as an inhibitor of BMP signaling. In addition, knockdown of both genes leads to a significant reduction/loss of the post-otic cranial neural crest. This results in a subsequent absence of neural crest precursors in the posterior pharyngeal arches and a loss of enteric precursors in the intestine.

Genes / Markers

Figures

Show all Figures

Expression

Gene	Fish	Conditions	Stage	Qualifier	Anatomy	Assay	Figure
crestin	WT	control	Prim-25		presumptive enteric nervous system	ISH	Fig. 3
crestin	WT + MO2-zeb2a	standard conditions	Prim-25		presumptive enteric nervous system	ISH	Fig. 3
crestin	WT + MO2-zeb2a + MO2-zeb2b	standard conditions	Prim-25		presumptive enteric nervous system	ISH	Fig. 3
crestin	WT + MO2-zeb2b	standard conditions	Prim-25		presumptive enteric nervous system	ISH	Fig. 3
dlx2a	WT	control	Long-pec		pharyngeal arch	ISH	Fig. 3
dlx2a	WT + MO2-zeb2a	standard conditions	Long-pec		pharyngeal arch	ISH	Fig. 3
dlx2a	WT + MO2-zeb2a	standard conditions	Long-pec	Not Detected	pharyngeal arch 3-7	ISH	Fig. 3
dlx2a	WT + MO2-zeb2a + MO2-zeb2b	standard conditions	Long-pec		pharyngeal arch	ISH	Fig. 3
dlx2a	WT + MO2-zeb2b	standard conditions	Long-pec		pharyngeal arch	ISH	Fig. 3
dlx2a	WT + MO2-zeb2b	standard conditions	Long-pec	Not Detected	pharyngeal arch 3-7	ISH	Fig. 3

1 - 10 of 90

Show

Phenotype

Fish	Conditions	Stage	Phenotype	Figure
WT + MO1-zeb2a	standard conditions	5-9 somites	dorsal convergence disrupted, abnormal	Fig. 3
WT + MO1-zeb2a	standard conditions	Prim-5 to Protruding-mouth	brain morphology, abnormal	text only
WT + MO1-zeb2a	standard conditions	Prim-5 to Protruding-mouth	central nervous system development disrupted, abnormal	text only
WT + MO1-zeb2a	standard conditions	Prim-5 to Protruding-mouth	heart morphology, abnormal	text only
WT + MO1-zeb2a	standard conditions	Prim-5 to Protruding-mouth	post-anal tail morphogenesis disrupted, abnormal	text only
WT + MO1-zeb2a	standard conditions	Prim-5 to Protruding-mouth	whole organism anterior-posterior axis curved ventral, abnormal	text only
WT + MO1-zeb2a + MO1-zeb2b	standard conditions	5-9 somites	convergent extension involved in axis elongation disrupted, abnormal	Fig. 3
WT + MO1-zeb2a + MO1-zeb2b	standard conditions	5-9 somites	dorsal convergence disrupted, abnormal	Fig. 3
WT + MO1-zeb2a + MO1-zeb2b	standard conditions	5-9 somites	somite morphology, abnormal	Fig. 3
WT + MO1-zeb2a + MO1-zeb2b	standard conditions	5-9 somites	whole organism anterior-posterior axis decreased length, abnormal	Fig. 3

1 - 10 of 69

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Mutations / Transgenics

Human Disease / Model

Sequence Targeting Reagents

Target	Reagent	Reagent Type
zeb2a	MO1-zeb2a	MRPHLNO
zeb2a	MO2-zeb2a	MRPHLNO
zeb2b	MO1-zeb2b	MRPHLNO
zeb2b	MO2-zeb2b	MRPHLNO

Fish

Fish
WT
WT + MO1-zeb2a
WT + MO1-zeb2a + MO1-zeb2b
WT + MO1-zeb2b
WT + MO2-zeb2a
WT + MO2-zeb2a + MO2-zeb2b
WT + MO2-zeb2b

Antibodies

Orthology

Gene	Orthology
sbds
zeb2b

Engineered Foreign Genes

Mapping

Delalande et al., 2008 ZDB-PUB-080327-2 PMID:18351671

Zebrafish sip1a and sip1b are essential for normal axial and neural patterning

Delalande et al., 2008

ZDB-PUB-080327-2
PMID:18351671