PUBLICATION

A role of glypican4 and wnt5b in chondrocyte stacking underlying craniofacial cartilage morphogenesis

Authors

Sisson, B.E., Dale, R.M., Mui, S.R., Topczewska, J.M., Topczewski, J.

ID

ZDB-PUB-151016-16

Date

2015

Source

Mechanisms of Development 138 Pt 3: 279-90 (Journal)

Registered Authors

Dale, Rodney M., Mui, Stephanie, Sisson, Barbara E., Topczewska, Jolanta, Topczewski, Jacek

Keywords

Growth plate, Wnt/PCP pathway, knypek, pipe tail

MeSH Terms

Glypicans/deficiency
Glypicans/genetics*
Wnt-5a Protein
Animals, Genetically Modified
Mutation
Cell Size
Zebrafish/embryology*
Zebrafish/genetics*
Zebrafish/metabolism
Gastrulation/genetics
Phenotype
Chondrogenesis/genetics*
Wnt Signaling Pathway/genetics
Cell Count
Neural Crest/embryology
Neural Crest/metabolism
Animals
Cell Movement/genetics
Chondrocytes/cytology
Chondrocytes/metabolism*
Zebrafish Proteins/deficiency
Zebrafish Proteins/genetics*
Wnt Proteins/deficiency
Wnt Proteins/genetics*
Branchial Region/embryology
Branchial Region/metabolism
Gene Expression Regulation, Developmental

(all 27)

PubMed

26459057 Full text @ Mech. Dev.

Abstract

The Wnt/Planar Cell Polarity (PCP) pathway controls cell morphology and behavior during animal development. Several zebrafish mutants were identified as having perturbed Wnt/PCP signaling. Many of these mutants have defects in craniofacial formation. To better understand the role that Wnt/PCP plays in craniofacial development we set out to identify which of the mutants, known to be associated with the Wnt/PCP pathway, perturb head cartilage formation by disrupting chondrocyte morphology. Here we demonstrate that while vang-like 2 (vangl2), wnt11 and scribbled (scrib) mutants have severe craniofacial morphogenesis defects they do not display the chondrocyte stacking and intercalation problems seen in glypican 4 (gpc4) and wnt5b mutants. The function of Gpc4 or Wnt5b appears to be important for chondrocyte organization, as the neural crest in both mutants is specified, undergoes migration, and differentiates into the same number of cells to compose the craniofacial cartilage elements. We demonstrate that Gpc4 activity is required cell autonomously in the chondrocytes and that the phenotype of single heterozygous mutants is slightly enhanced in embryos double heterozygous for wnt5b and gpc4. This data suggests a novel mechanism for Wnt5b and Gpc4 regulation of chondrocyte behavior that is independent of the core Wnt/PCP molecules and differs from their collaborative action of controlling cell movements during gastrulation.

Genes / Markers

Figures

Show all Figures

Expression

Gene	Fish	Conditions	Stage	Anatomy	Assay	Figure
dlx2a	gpc4^fr6/fr6	control	Prim-5	pharyngeal arch	ISH	Fig. 3
dlx2a	wnt5b^ta98/ta98	control	Prim-5	pharyngeal arch	ISH	Fig. 3
dlx2a	WT	control	Prim-5	pharyngeal arch	ISH	Fig. 3
EGFP	ba2Tg	control	Long-pec	pharyngeal arch	IFL	Fig. 1
foxd3	gpc4^fr6/fr6	standard conditions	5-9 somites	premigratory neural crest	ISH	Fig. 3
foxd3	wnt5b^ta98/ta98	standard conditions	5-9 somites	premigratory neural crest	ISH	Fig. 3
foxd3	WT	standard conditions	5-9 somites	premigratory neural crest	ISH	Fig. 3
gpc4	AB	control	Protruding-mouth	pharyngeal arch	ISH	Fig. 1
gpc4	AB	control	Long-pec	pharyngeal arch	ISH	Fig. 1
hand2	gpc4^fr6/fr6	control	Prim-15	pharyngeal arch	ISH	Fig. 3

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Phenotype

Fish	Conditions	Stage	Phenotype	Figure
gpc4^fr6/fr6	control	Prim-5	neural crest cell fate specification process quality, normal	Fig. 3
gpc4^fr6/fr6	control	Prim-15	neural crest cell fate specification process quality, normal	Fig. 3
gpc4^fr6/fr6	standard conditions	5-9 somites	convergent extension process quality, abnormal	Fig. 3
gpc4^fr6/fr6	standard conditions	5-9 somites	neural crest cell fate specification process quality, normal	Fig. 3
gpc4^fr6/fr6	standard conditions	Day 4	ceratohyal cartilage chondrocyte amount, normal	Fig. 5
gpc4^fr6/fr6	standard conditions	Day 4	ceratohyal cartilage chondrocyte circular, abnormal	Fig. 2
gpc4^fr6/fr6	standard conditions	Day 4	ceratohyal cartilage chondrocyte decreased size, abnormal	Fig. 5
gpc4^fr6/fr6	standard conditions	Day 4	ceratohyal cartilage condensed, abnormal	Fig. 2
gpc4^fr6/fr6	standard conditions	Day 4	ceratohyal cartilage decreased length, abnormal	Fig. 5
gpc4^fr6/fr6	standard conditions	Day 4	ceratohyal cartilage decreased volume, abnormal	Fig. 5

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Mutations / Transgenics

Allele	Construct	Type	Affected Genomic Region
ba2Tg	Tg(-4.9sox10:EGFP)	Transgenic Insertion
fr6		Point Mutation	gpc4
m209		Point Mutation	vangl2
m818		Point Mutation	gpc4
rw468		Point Mutation	scrib
ta98		Point Mutation	wnt5b
tz216		Point Mutation	wnt11f2
y1Tg	Tg(fli1:EGFP)	Transgenic Insertion

1 - 8 of 8

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Human Disease / Model

Sequence Targeting Reagents

Fish

Fish
AB
ba2Tg
gpc4^fr6/fr6
gpc4^fr6/fr6; y1Tg
gpc4^m818/m818
gpc4^m818/m818; wnt5b^ta98/+
gpc4^m818/m818; wnt5b^ta98/ta98
gpc4^m818/+
gpc4^m818/+; wnt5b^ta98/+
gpc4^m818/+; wnt5b^ta98/ta98

Antibodies

Orthology

Engineered Foreign Genes

Marker	Marker Type	Name
EGFP	EFG	EGFP

Mapping

Sisson et al., 2015 ZDB-PUB-151016-16 PMID:26459057

A role of glypican4 and wnt5b in chondrocyte stacking underlying craniofacial cartilage morphogenesis

Sisson et al., 2015

ZDB-PUB-151016-16
PMID:26459057