PUBLICATION

Pdgfra and Pdgfrb genetically interact during craniofacial development

Authors

McCarthy, N., Liu, J.S., Richarte, A.M., Eskiocak, B., Lovely, C.B., Tallquist, M.D., Eberhart, J.K.

ID

ZDB-PUB-160315-9

Date

2016

Source

Developmental Dynamics : an official publication of the American Association of Anatomists 245(6): 641-52 (Journal)

Registered Authors

Eberhart, Johann, Lovely, Ben, McCarthy, Neil

Keywords

cranial neural crest, palatogenesis, pdgfra, pdgfrb

MeSH Terms

Female
Mice
Signal Transduction/genetics
Signal Transduction/physiology
Immunohistochemistry
Animals
Male
Receptor, Platelet-Derived Growth Factor beta/genetics
Receptor, Platelet-Derived Growth Factor beta/metabolism*
Neural Crest/embryology
Neural Crest/metabolism
Gene Expression Regulation, Developmental/genetics
Gene Expression Regulation, Developmental/physiology
Receptor, Platelet-Derived Growth Factor alpha/genetics
Receptor, Platelet-Derived Growth Factor alpha/metabolism*
Mice, Inbred C57BL
Zebrafish Proteins/genetics
Zebrafish Proteins/metabolism
Cleft Palate/embryology
Cleft Palate/genetics
Cleft Palate/metabolism
In Situ Hybridization
Zebrafish/embryology
Zebrafish/genetics

(all 24)

PubMed

26971580 Full text @ Dev. Dyn.

Abstract

Background One of the most prevalent congenital birth defects is cleft palate. The palatal skeleton is derived from the cranial neural crest and platelet-derived growth factors (Pdgf) are critical in palatogenesis. Of the two Pdgf receptors, pdgfra is required for neural crest migration and palatogenesis. However, the role pdgfrb plays in the neural crest, or whether pdgfra and pdgfrb interact during palatogenesis is unclear.

Results We find that pdgfrb is dispensable for craniofacial development in zebrafish. However, the palatal defect in pdgfra;pdgfrb double mutants is significantly more severe than in pdgfra single mutants. Data in mouse suggest this interaction is conserved and that neural crest requires both genes. In zebrafish, pdgfra and pdgfrb are both expressed by neural crest within the pharyngeal arches and pharmacological analyses demonstrate Pdgf signaling is required at these times. While neither proliferation nor cell death appears affected, time-lapsed confocal analysis of pdgfra;pdgfrb mutants shows a failure of proper neural crest condensation during palatogenesis.

Conclusions We provide data showing that pdgfra and pdgfrb interact during palatogenesis in both zebrafish and mouse. In zebrafish, this interaction affects proper condensation of maxillary neural crest cells, revealing a previously unknown interaction between Pdgfra and Pdgfrb during palate formation. This article is protected by copyright. All rights reserved.

Genes / Markers

Figures

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Expression

Gene	Fish	Conditions	Stage	Anatomy	Assay	Figure
EGFP	pdgfra^b1059/b1059; pdgfrb^um148/um148; y1Tg	standard conditions	Prim-15	chondrocranium cartilage cranial neural crest cell	IFL	Fig. 7
EGFP	pdgfra^b1059/b1059; pdgfrb^um148/um148; y1Tg	standard conditions	Prim-5	chondrocranium cartilage cranial neural crest cell	IFL	Fig. 8
EGFP	pdgfra^b1059/b1059; pdgfrb^um148/um148; y1Tg	standard conditions	Prim-5	chondrocranium cartilage cranial neural crest cell	IHC	Fig. 10
EGFP	pdgfra^b1059/b1059; pdgfrb^um148/um148; y1Tg	standard conditions	Prim-25	chondrocranium cartilage cranial neural crest cell	IFL	Fig. 7
EGFP	pdgfra^b1059/b1059; pdgfrb^um148/um148; y1Tg	standard conditions	Prim-15	chondrocranium cartilage cranial neural crest cell	IFL	Fig. 9
EGFP	pdgfra^b1059/b1059; pdgfrb^um148/um148; y1Tg	standard conditions	Prim-5	chondrocranium cartilage cranial neural crest cell	IFL	Fig. 7
EGFP	pdgfra^b1059/b1059; y1Tg	standard conditions	Prim-15	chondrocranium cartilage cranial neural crest cell	IFL	Fig. 7
EGFP	pdgfra^b1059/b1059; y1Tg	standard conditions	Prim-25	chondrocranium cartilage cranial neural crest cell	IFL	Fig. 7
EGFP	pdgfra^b1059/b1059; y1Tg	standard conditions	Prim-5	chondrocranium cartilage cranial neural crest cell	IFL	Fig. 7
EGFP	pdgfra^b1059/b1059; y1Tg	standard conditions	Prim-15	chondrocranium cartilage cranial neural crest cell	IFL	Fig. 9

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Phenotype

Fish	Conditions	Stage	Phenotype	Figure
WT	chemical treatment by environment: Ki11502	Day 5	embryonic neurocranium morphogenesis decreased process quality, abnormal	Fig. 4
WT	chemical treatment by environment: Ki11502	Day 5	neurocranium lacks all parts of type ethmoid cartilage, abnormal	Fig. 4
WT	chemical treatment by environment: Ki11502	Day 5	neurocranium lacks all parts of type neurocranial trabecula, abnormal	Fig. 4
WT	chemical treatment by environment: Ki11502	Day 5	roof of mouth development decreased process quality, abnormal	Fig. 4
pdgfra^b1059/+	standard conditions	Day 5	embryonic neurocranium morphogenesis process quality, normal	Fig. 1
pdgfra^b1059/+	standard conditions	Day 5	neurocranium length, normal	Fig. 1
pdgfra^b1059/+	standard conditions	Day 5	neurocranium morphology, normal	Fig. 1
pdgfra^b1059/+; pdgfrb^um148/+	standard conditions	Day 5	embryonic neurocranium morphogenesis process quality, normal	Fig. 1
pdgfra^b1059/+; pdgfrb^um148/+	standard conditions	Day 5	neurocranium length, normal	Fig. 1
pdgfra^b1059/+; pdgfrb^um148/+	standard conditions	Day 5	neurocranium morphology, normal	Fig. 1

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Mutations / Transgenics

Allele	Construct	Type	Affected Genomic Region
b1059		Point Mutation	pdgfra
um148		Small Deletion	pdgfrb
y1Tg	Tg(fli1:EGFP)	Transgenic Insertion

Human Disease / Model

Human Disease	Fish	Conditions	Evidence
cleft palate			TAS

Sequence Targeting Reagents

Fish

Fish
pdgfra^b1059/b1059
pdgfra^b1059/b1059; pdgfrb^um148/+
pdgfra^b1059/b1059; pdgfrb^um148/um148
pdgfra^b1059/b1059; pdgfrb^um148/um148; y1Tg
pdgfra^b1059/b1059; y1Tg
pdgfra^b1059/+
pdgfra^b1059/+; pdgfrb^um148/+
pdgfra^b1059/+; pdgfrb^um148/um148
pdgfrb^um148/+
pdgfrb^um148/um148

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Antibodies

Orthology

Engineered Foreign Genes

Marker	Marker Type	Name
EGFP	EFG	EGFP

Mapping

McCarthy et al., 2016 ZDB-PUB-160315-9 PMID:26971580

Pdgfra and Pdgfrb genetically interact during craniofacial development

McCarthy et al., 2016

ZDB-PUB-160315-9
PMID:26971580