PUBLICATION

Mutations in cdon and boc affect trunk neural crest cell migration and slow-twitch muscle development in zebrafish

Authors

Lencer, E., Rains, A., Binne, E., Prekeris, R., Artinger, K.

ID

ZDB-PUB-230701-32

Date

2023

Source

Development (Cambridge, England) 150(14): (Journal)

Registered Authors

Artinger, Kristin Bruk, Lencer, Ezra

Keywords

Adaxial Cell, Cell Guidance, Cell Migration, Extracellular Matrix, Hedghog Signaling, Neural Crest Cell

MeSH Terms

Cell Differentiation
Mutation/genetics
Cell Movement/genetics
Neural Crest*
Cell Adhesion Molecules/genetics
Zebrafish*/genetics
Zebrafish*/metabolism
Muscle Development/genetics
Animals
Hedgehog Proteins/genetics
Hedgehog Proteins/metabolism

PubMed

37390228 Full text @ Development

Abstract

The transmembrane proteins cdon and boc are implicated in regulating hedgehog signaling during vertebrate development. Recent work showing roles for these genes in axon guidance and neural crest cell migration suggest that cdon/boc may play additional functions in regulating directed cell movements. We use novel and existing mutants to investigate a role for cdon and boc in zebrafish neural crest cell migration. We find that single mutant embryos exhibit normal neural crest phenotypes, but that neural crest migration is strikingly disrupted in double cdon;boc mutant embryos. We further show that this migration phenotype is associated with defects to the differentiation of slow-twitch muscle cells, and the loss of a Col1a1a containing extracellular matrix, suggesting that neural crest defects may be a secondary consequence to defects in mesoderm development. Combined, our data add to a growing literature showing that cdon and boc act synergistically to promote hedgehog signaling during vertebrate development, and suggest that the zebrafish can be used to study the function of hedgehog receptor paralogs.

Genes / Markers

Figures

Show all Figures

Expression

Fish	Conditions	Stage	Qualifier	Anatomy	Assay	Figure
i106Tg	standard conditions	1-4 somites	Not Detected	adaxial cell	ISH	Fig. 2
i106Tg	standard conditions	10-13 somites		mesoderm	ISH	Fig. 2
i106Tg	standard conditions	Prim-5		slow muscle cell	ISH	Fig. 2
vu234Tg	standard conditions	Prim-5		neural crest cell	ISH	Fig. 2
WT	standard conditions	Long-pec		central nervous system	ISH	Fig. 2
WT	standard conditions	1-4 somites		neural plate border	ISH	Fig. 2
WT	standard conditions	Prim-5		neural tube	ISH	Fig. 2
i106Tg	standard conditions	1-4 somites	Not Detected	adaxial cell	ISH	Fig. 2
i106Tg	standard conditions	10-13 somites		mesoderm	ISH	Fig. 2
i106Tg	standard conditions	Prim-5		slow muscle cell	ISH	Fig. 2

1 - 10 of 16

Show

Phenotype

Fish	Conditions	Stage	Phenotype	Figure
boc^co24/co24	chemical treatment by environment: Cyclopamine	Prim-5	slow muscle cell decreased amount, exacerbated	Fig. 4
boc^co24/co24; st1002Tg	standard conditions	Prim-5	neural tube Wnt signaling pathway decreased occurrence, abnormal	Fig. 3
boc^co24/co24; st1002Tg	standard conditions	Prim-5	slow muscle cell Wnt signaling pathway decreased occurrence, abnormal	Fig. 3
cdon^{co1019/co1019}	standard conditions	Prim-5	slow muscle cell ab-f59 labeling decreased amount, abnormal	Fig. 3
cdon^{co1019/co1019}	chemical treatment by environment: Cyclopamine	Prim-5	slow muscle cell decreased amount, abnormal	Fig. 4
cdon^{co1019/co1019}; boc^co24/co24	control	Prim-5	slow muscle cell decreased amount, abnormal	Fig. 4
cdon^{co1019/co1019}; boc^co24/co24	standard conditions	Prim-5	slow muscle cell ab-f59 labeling decreased amount, abnormal	Fig. 3
cdon^{co1019/co1019}; boc^co24/co24	chemical treatment by environment: Cyclopamine	Prim-5	slow muscle cell decreased amount, exacerbated	Fig. 4
cdon^{co1019/co1019}; boc^co24/co24; st1002Tg	standard conditions	Prim-5	neural tube Wnt signaling pathway decreased occurrence, abnormal	Fig. 3
cdon^{co1019/co1019}; boc^co24/co24; st1002Tg	standard conditions	Prim-5	slow muscle cell Wnt signaling pathway decreased occurrence, abnormal	Fig. 3

1 - 10 of 13

Show

Mutations / Transgenics

Allele	Construct	Type	Affected Genomic Region
co24		Unknown	boc
co1019		Small Deletion	cdon
co1023		Small Deletion	cdon
i106Tg	TgPAC(prdm1a:EGFP)	Transgenic Insertion
st1002Tg	Tg(8xgli-Xla.Cryaa:NLS-d1mCherry)	Transgenic Insertion
ty54z		Point Mutation	boc
vu234Tg	Tg(sox10:mRFP)	Transgenic Insertion

1 - 7 of 7

Show

Human Disease / Model

Sequence Targeting Reagents

Fish

Fish
boc^co24/co24
boc^co24/co24; st1002Tg
boc^co24/co24; vu234Tg
cdon^{co1019/co1019}
cdon^{co1019/co1019}; boc^co24/co24
cdon^{co1019/co1019}; boc^co24/co24; st1002Tg
cdon^{co1019/co1019}; boc^co24/co24; vu234Tg
cdon^{co1019/co1019}; st1002Tg
cdon^{co1019/co1019}; vu234Tg
i106Tg

1 - 10 of 14

Show

Antibodies

Orthology

Engineered Foreign Genes

Marker	Marker Type	Name
EGFP	EFG	EGFP
mCherry	EFG	mCherry
mRFP	EFG	mRFP

Mapping

Lencer et al., 2023 ZDB-PUB-230701-32 PMID:37390228

Mutations in cdon and boc affect trunk neural crest cell migration and slow-twitch muscle development in zebrafish

Lencer et al., 2023

ZDB-PUB-230701-32
PMID:37390228