PUBLICATION

Crispld2 is required for neural crest cell migration and cell viability during zebrafish craniofacial development

Authors

Swindell, E.C., Yuan, Q., Maili, L.E., Tandon, B., Wagner, D.S., Hecht, J.T.

ID

ZDB-PUB-150825-57

Date

2015

Source

Genesis (New York, N.Y. : 2000) 53(10): 660-7 (Journal)

Registered Authors

Swindell, Eric C., Wagner, Daniel

Keywords

Birth Defects, Early Development, Migration, Neural Crest, Other

MeSH Terms

Cell Differentiation/genetics
Zebrafish Proteins/genetics*
Zebrafish Proteins/metabolism
Humans
Neural Crest/cytology
Neural Crest/embryology
Neural Crest/metabolism*
Time Factors
Apoptosis/genetics
Skull/embryology
Skull/metabolism
Fluorescent Antibody Technique
Animals
Cell Survival/genetics
Time-Lapse Imaging
RNA, Messenger/genetics
RNA, Messenger/metabolism
Microscopy, Confocal
Head/embryology
Gene Knockdown Techniques
Body Patterning/genetics
Glycoproteins/genetics*
Glycoproteins/metabolism
Gene Expression Regulation, Developmental
Cell Movement/genetics*
Zebrafish/embryology
Zebrafish/genetics*
Zebrafish/metabolism

(all 28)

PubMed

26297922 Full text @ Genesis

Abstract

The CAP superfamily member, CRISPLD2, has previously been shown to be associated with nonsyndromic cleft lip and palate (NSCL/P) in human populations and to be essential for normal craniofacial development in the zebrafish. Additionally, in rodent models, CRISPLD2 has been shown to play a role in normal lung and kidney development. However, the specific role of CRISPLD2 during these developmental processes has yet to be determined. In this study, we demonstrate that Crispld2 protein localizes to the orofacial region of the zebrafish embryo and knockdown of crispld2 results in abnormal migration of neural crest cells during both early and late time points. We also show an increase in cell death after crispld2 knockdown as well as an increase in apoptotic marker genes. Our data suggests that Crispld2 modulates the migration, differentiation and/or survival of NCCs during early craniofacial development. These results indicate an important role for Crispld2 in neural crest cell migration during craniofacial development and suggests involvement of Crispld2 in cell viability during formation of the orofacies. This article is protected by copyright. All rights reserved.

Genes / Markers

Figures

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Expression

Gene	Antibody	Fish	Conditions	Stage	Anatomy	Assay	Figure
casp8		WT	standard conditions	14-19 somites	whole organism	RTPCR	Fig. 6
casp8		WT + MO3-crispld2	standard conditions	14-19 somites	whole organism	RTPCR	Fig. 6
crispld2	ab1-crispld2	WT	standard conditions	Long-pec	pharynx	IHC	Fig. 5
EGFP		ba2Tg	standard conditions	14-19 somites	neural crest cell	IFL	Fig. 1
EGFP		ba2Tg	standard conditions	26+ somites	neural crest cell	IFL	Fig. 1
EGFP		ba2Tg	standard conditions	Prim-5	neural crest cell	IFL	Fig. 1
EGFP		ba2Tg	standard conditions	Protruding-mouth	pharyngeal arch cartilage	IFL	Fig. 2
EGFP		ba2Tg	standard conditions	Pec-fin	pharyngeal arch cartilage	IFL	Fig. 2
EGFP		ba2Tg	standard conditions	Long-pec	pharyngeal arch cartilage	IFL	Fig. 2
EGFP		ba2Tg + MO3-crispld2	standard conditions	14-19 somites	neural crest cell	IFL	Fig. 1

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Phenotype

Fish	Conditions	Stage	Phenotype	Figure
WT + MO3-crispld2	standard conditions	14-19 somites	head apoptotic process increased occurrence, abnormal	Fig. 3
WT + MO3-crispld2	standard conditions	14-19 somites	head cell population proliferation occurrence, normal	Fig. 4
WT + MO3-crispld2	standard conditions	14-19 somites	whole organism casp8 expression increased amount, abnormal	Fig. 6
WT + MO3-crispld2	standard conditions	14-19 somites	whole organism mmp2 expression increased amount, abnormal	Fig. 6
WT + MO3-crispld2	standard conditions	Prim-5	head apoptotic process increased occurrence, abnormal	Fig. 3
WT + MO3-crispld2	standard conditions	Prim-5	head cell population proliferation occurrence, normal	Fig. 4
WT + MO3-crispld2	standard conditions	Prim-5	pharyngeal arch 3-7 cell population proliferation decreased occurrence, abnormal	Fig. 4
WT + MO3-crispld2	standard conditions	Prim-25	head apoptotic process increased occurrence, abnormal	Fig. 3
WT + MO3-crispld2	standard conditions	Long-pec	head apoptotic process increased occurrence, abnormal	Fig. 3
WT + MO3-crispld2	standard conditions	Long-pec	head cell population proliferation occurrence, normal	Fig. 4

1 - 10 of 24

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Mutations / Transgenics

Allele	Construct	Type	Affected Genomic Region
ba2Tg	Tg(-4.9sox10:EGFP)	Transgenic Insertion

Human Disease / Model

Sequence Targeting Reagents

Target	Reagent	Reagent Type
crispld2	MO3-crispld2	MRPHLNO
tp53	MO4-tp53	MRPHLNO

Fish

Fish
ba2Tg
ba2Tg + MO3-crispld2
WT
WT + MO3-crispld2
WT + MO3-crispld2 + MO4-tp53

Antibodies

Name	Type	Antigen Genes	Isotypes	Host Organism
Ab1-crispld2		crispld2		Rabbit

Orthology

Engineered Foreign Genes

Marker	Marker Type	Name
EGFP	EFG	EGFP

Mapping

Swindell et al., 2015 ZDB-PUB-150825-57 PMID:26297922

Crispld2 is required for neural crest cell migration and cell viability during zebrafish craniofacial development

Swindell et al., 2015

ZDB-PUB-150825-57
PMID:26297922