Early craniofacial defects in zebrafish that have reduced function of a wnt-interacting extracellular matrix protein, Tinagl1

Hannah Neiswender, Sammy Navarre, David J Kozlowski, Ellen LeMosy

Research output: Contribution to journalArticle

1 Citation (Scopus)

Abstract

Objective: Tinagl1 has a weak genetic association with craniosynostosis, but its functions in cartilage and bone development are unknown. Knockdown of Tinagl1 in zebrafish embryos allowed an initial characterization of its potential effects on craniofacial cartilage development and a test of whether these effects could involve Wnt signaling. Results: Tinagl1 knockdown resulted in dose-dependent reductions and defects in ventral pharyngeal arch cartilages as well as the ethmoid plate, a zebrafish correlate to the palate. These defects could be correlated to reduced numbers of cranial neural crest cells in the pharyngeal arches and could be reproduced with comanipulation of Tinagl1 and Wnt3a by morpholino-based knockdown. Conclusions: These results suggest that Tinagl1 is required early in the proliferation or migration of cranial neural crest cells and that its effects are mediated via Wnt3a signaling. Because Wnt3a is among the Wnts that contribute to nonsyndromic cleft lip and cleft palate in mouse and man, further investigation of Tinagl1 may help to elucidate mechanisms underlying these disorders.

Original languageEnglish (US)
Pages (from-to)381-390
Number of pages10
JournalCleft Palate-Craniofacial Journal
Volume54
Issue number4
DOIs
StatePublished - May 1 2017

Fingerprint

Extracellular Matrix Proteins
Zebrafish
Cartilage
Branchial Region
Neural Crest
Morpholinos
Craniosynostoses
Palate
Bone Development
Cleft Palate
Embryonic Structures

Keywords

  • Dlx2a
  • Neural crest cells
  • Tinagl1
  • Wnt3A
  • Zebrafish

ASJC Scopus subject areas

  • Oral Surgery
  • Otorhinolaryngology

Cite this

Early craniofacial defects in zebrafish that have reduced function of a wnt-interacting extracellular matrix protein, Tinagl1. / Neiswender, Hannah; Navarre, Sammy; Kozlowski, David J; LeMosy, Ellen.

In: Cleft Palate-Craniofacial Journal, Vol. 54, No. 4, 01.05.2017, p. 381-390.

Research output: Contribution to journalArticle

@article{3896d66e5f0e4461ba3cb1723008a8cd,
title = "Early craniofacial defects in zebrafish that have reduced function of a wnt-interacting extracellular matrix protein, Tinagl1",
abstract = "Objective: Tinagl1 has a weak genetic association with craniosynostosis, but its functions in cartilage and bone development are unknown. Knockdown of Tinagl1 in zebrafish embryos allowed an initial characterization of its potential effects on craniofacial cartilage development and a test of whether these effects could involve Wnt signaling. Results: Tinagl1 knockdown resulted in dose-dependent reductions and defects in ventral pharyngeal arch cartilages as well as the ethmoid plate, a zebrafish correlate to the palate. These defects could be correlated to reduced numbers of cranial neural crest cells in the pharyngeal arches and could be reproduced with comanipulation of Tinagl1 and Wnt3a by morpholino-based knockdown. Conclusions: These results suggest that Tinagl1 is required early in the proliferation or migration of cranial neural crest cells and that its effects are mediated via Wnt3a signaling. Because Wnt3a is among the Wnts that contribute to nonsyndromic cleft lip and cleft palate in mouse and man, further investigation of Tinagl1 may help to elucidate mechanisms underlying these disorders.",
keywords = "Dlx2a, Neural crest cells, Tinagl1, Wnt3A, Zebrafish",
author = "Hannah Neiswender and Sammy Navarre and Kozlowski, {David J} and Ellen LeMosy",
year = "2017",
month = "5",
day = "1",
doi = "10.1597/15-283",
language = "English (US)",
volume = "54",
pages = "381--390",
journal = "Cleft Palate-Craniofacial Journal",
issn = "1055-6656",
publisher = "American Cleft Palate Craniofacial Association",
number = "4",

}

TY - JOUR

T1 - Early craniofacial defects in zebrafish that have reduced function of a wnt-interacting extracellular matrix protein, Tinagl1

AU - Neiswender, Hannah

AU - Navarre, Sammy

AU - Kozlowski, David J

AU - LeMosy, Ellen

PY - 2017/5/1

Y1 - 2017/5/1

N2 - Objective: Tinagl1 has a weak genetic association with craniosynostosis, but its functions in cartilage and bone development are unknown. Knockdown of Tinagl1 in zebrafish embryos allowed an initial characterization of its potential effects on craniofacial cartilage development and a test of whether these effects could involve Wnt signaling. Results: Tinagl1 knockdown resulted in dose-dependent reductions and defects in ventral pharyngeal arch cartilages as well as the ethmoid plate, a zebrafish correlate to the palate. These defects could be correlated to reduced numbers of cranial neural crest cells in the pharyngeal arches and could be reproduced with comanipulation of Tinagl1 and Wnt3a by morpholino-based knockdown. Conclusions: These results suggest that Tinagl1 is required early in the proliferation or migration of cranial neural crest cells and that its effects are mediated via Wnt3a signaling. Because Wnt3a is among the Wnts that contribute to nonsyndromic cleft lip and cleft palate in mouse and man, further investigation of Tinagl1 may help to elucidate mechanisms underlying these disorders.

AB - Objective: Tinagl1 has a weak genetic association with craniosynostosis, but its functions in cartilage and bone development are unknown. Knockdown of Tinagl1 in zebrafish embryos allowed an initial characterization of its potential effects on craniofacial cartilage development and a test of whether these effects could involve Wnt signaling. Results: Tinagl1 knockdown resulted in dose-dependent reductions and defects in ventral pharyngeal arch cartilages as well as the ethmoid plate, a zebrafish correlate to the palate. These defects could be correlated to reduced numbers of cranial neural crest cells in the pharyngeal arches and could be reproduced with comanipulation of Tinagl1 and Wnt3a by morpholino-based knockdown. Conclusions: These results suggest that Tinagl1 is required early in the proliferation or migration of cranial neural crest cells and that its effects are mediated via Wnt3a signaling. Because Wnt3a is among the Wnts that contribute to nonsyndromic cleft lip and cleft palate in mouse and man, further investigation of Tinagl1 may help to elucidate mechanisms underlying these disorders.

KW - Dlx2a

KW - Neural crest cells

KW - Tinagl1

KW - Wnt3A

KW - Zebrafish

UR - http://www.scopus.com/inward/record.url?scp=85023204644&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=85023204644&partnerID=8YFLogxK

U2 - 10.1597/15-283

DO - 10.1597/15-283

M3 - Article

VL - 54

SP - 381

EP - 390

JO - Cleft Palate-Craniofacial Journal

JF - Cleft Palate-Craniofacial Journal

SN - 1055-6656

IS - 4

ER -