Latent TGF-beta binding protein-1 plays an important role in craniofacial development
Autor(a) principal: | |
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Data de Publicação: | 2020 |
Outros Autores: | , , , |
Tipo de documento: | Artigo |
Idioma: | eng |
Título da fonte: | Journal of applied oral science (Online) |
Texto Completo: | http://old.scielo.br/scielo.php?script=sci_arttext&pid=S1678-77572020000100481 |
Resumo: | Abstract Objective: This study aims to replicate the phenotype of Ltbp1 knockout mice in zebrafish, and to address the function of LTBP1 in craniofacial development. Methods: Whole mount in situ hybridization (WISH) of ltbp1 was performed at critical periods of zebrafish craniofacial development to explore the spatial-temporal expression pattern. Furthermore, we generated morpholino based knockdown model of ltbp1 to study the craniofacial phenotype. Results: WISH of ltbp1 was mainly detected in the mandibular jaw region, brain trunk, and internal organs such as pancreas and gallbladder. And ltbp1 colocalized with both sox9a and ckma in mandibular region. Morpholino based knockdown of ltbp1 results in severe jaw malformation. Alcian blue staining revealed severe deformity of Meckel's cartilage along with the absence of ceratobranchial. Three-dimension measurements of ltbp1 morphants jaws showed decrease in both mandible length and width and increase in open mouth distance. Expression of cartilage marker sox9a and muscle marker ckma was decreased in ltbp1 morphants. Conclusions: Our experiments found that ltbp1 was expressed in zebrafish mandibular jaw cartilages and the surrounding muscles. The ltbp1 knockdown zebrafish exhibited phenotypes consistent with Ltbp1 knockout mice. And loss of ltbp1 function lead to significant mandibular jaw defects and affect both jaw cartilages and surrounding muscles. |
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Journal of applied oral science (Online) |
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Latent TGF-beta binding protein-1 plays an important role in craniofacial developmentLTBP1Craniofacial anomaliesDevelopmental biologyZebrafishAbstract Objective: This study aims to replicate the phenotype of Ltbp1 knockout mice in zebrafish, and to address the function of LTBP1 in craniofacial development. Methods: Whole mount in situ hybridization (WISH) of ltbp1 was performed at critical periods of zebrafish craniofacial development to explore the spatial-temporal expression pattern. Furthermore, we generated morpholino based knockdown model of ltbp1 to study the craniofacial phenotype. Results: WISH of ltbp1 was mainly detected in the mandibular jaw region, brain trunk, and internal organs such as pancreas and gallbladder. And ltbp1 colocalized with both sox9a and ckma in mandibular region. Morpholino based knockdown of ltbp1 results in severe jaw malformation. Alcian blue staining revealed severe deformity of Meckel's cartilage along with the absence of ceratobranchial. Three-dimension measurements of ltbp1 morphants jaws showed decrease in both mandible length and width and increase in open mouth distance. Expression of cartilage marker sox9a and muscle marker ckma was decreased in ltbp1 morphants. Conclusions: Our experiments found that ltbp1 was expressed in zebrafish mandibular jaw cartilages and the surrounding muscles. The ltbp1 knockdown zebrafish exhibited phenotypes consistent with Ltbp1 knockout mice. And loss of ltbp1 function lead to significant mandibular jaw defects and affect both jaw cartilages and surrounding muscles.Faculdade De Odontologia De Bauru - USP2020-01-01info:eu-repo/semantics/articleinfo:eu-repo/semantics/publishedVersiontext/htmlhttp://old.scielo.br/scielo.php?script=sci_arttext&pid=S1678-77572020000100481Journal of Applied Oral Science v.28 2020reponame:Journal of applied oral science (Online)instname:Universidade de São Paulo (USP)instacron:USP10.1590/1678-7757-2020-0262info:eu-repo/semantics/openAccessXiong,YitingSun,RongrongLi,JingyuWu,YueZhang,Jingjueng2020-11-25T00:00:00Zoai:scielo:S1678-77572020000100481Revistahttp://www.scielo.br/jaosPUBhttps://old.scielo.br/oai/scielo-oai.php||jaos@usp.br1678-77651678-7757opendoar:2020-11-25T00:00Journal of applied oral science (Online) - Universidade de São Paulo (USP)false |
dc.title.none.fl_str_mv |
Latent TGF-beta binding protein-1 plays an important role in craniofacial development |
title |
Latent TGF-beta binding protein-1 plays an important role in craniofacial development |
spellingShingle |
Latent TGF-beta binding protein-1 plays an important role in craniofacial development Xiong,Yiting LTBP1 Craniofacial anomalies Developmental biology Zebrafish |
title_short |
Latent TGF-beta binding protein-1 plays an important role in craniofacial development |
title_full |
Latent TGF-beta binding protein-1 plays an important role in craniofacial development |
title_fullStr |
Latent TGF-beta binding protein-1 plays an important role in craniofacial development |
title_full_unstemmed |
Latent TGF-beta binding protein-1 plays an important role in craniofacial development |
title_sort |
Latent TGF-beta binding protein-1 plays an important role in craniofacial development |
author |
Xiong,Yiting |
author_facet |
Xiong,Yiting Sun,Rongrong Li,Jingyu Wu,Yue Zhang,Jingju |
author_role |
author |
author2 |
Sun,Rongrong Li,Jingyu Wu,Yue Zhang,Jingju |
author2_role |
author author author author |
dc.contributor.author.fl_str_mv |
Xiong,Yiting Sun,Rongrong Li,Jingyu Wu,Yue Zhang,Jingju |
dc.subject.por.fl_str_mv |
LTBP1 Craniofacial anomalies Developmental biology Zebrafish |
topic |
LTBP1 Craniofacial anomalies Developmental biology Zebrafish |
description |
Abstract Objective: This study aims to replicate the phenotype of Ltbp1 knockout mice in zebrafish, and to address the function of LTBP1 in craniofacial development. Methods: Whole mount in situ hybridization (WISH) of ltbp1 was performed at critical periods of zebrafish craniofacial development to explore the spatial-temporal expression pattern. Furthermore, we generated morpholino based knockdown model of ltbp1 to study the craniofacial phenotype. Results: WISH of ltbp1 was mainly detected in the mandibular jaw region, brain trunk, and internal organs such as pancreas and gallbladder. And ltbp1 colocalized with both sox9a and ckma in mandibular region. Morpholino based knockdown of ltbp1 results in severe jaw malformation. Alcian blue staining revealed severe deformity of Meckel's cartilage along with the absence of ceratobranchial. Three-dimension measurements of ltbp1 morphants jaws showed decrease in both mandible length and width and increase in open mouth distance. Expression of cartilage marker sox9a and muscle marker ckma was decreased in ltbp1 morphants. Conclusions: Our experiments found that ltbp1 was expressed in zebrafish mandibular jaw cartilages and the surrounding muscles. The ltbp1 knockdown zebrafish exhibited phenotypes consistent with Ltbp1 knockout mice. And loss of ltbp1 function lead to significant mandibular jaw defects and affect both jaw cartilages and surrounding muscles. |
publishDate |
2020 |
dc.date.none.fl_str_mv |
2020-01-01 |
dc.type.driver.fl_str_mv |
info:eu-repo/semantics/article |
dc.type.status.fl_str_mv |
info:eu-repo/semantics/publishedVersion |
format |
article |
status_str |
publishedVersion |
dc.identifier.uri.fl_str_mv |
http://old.scielo.br/scielo.php?script=sci_arttext&pid=S1678-77572020000100481 |
url |
http://old.scielo.br/scielo.php?script=sci_arttext&pid=S1678-77572020000100481 |
dc.language.iso.fl_str_mv |
eng |
language |
eng |
dc.relation.none.fl_str_mv |
10.1590/1678-7757-2020-0262 |
dc.rights.driver.fl_str_mv |
info:eu-repo/semantics/openAccess |
eu_rights_str_mv |
openAccess |
dc.format.none.fl_str_mv |
text/html |
dc.publisher.none.fl_str_mv |
Faculdade De Odontologia De Bauru - USP |
publisher.none.fl_str_mv |
Faculdade De Odontologia De Bauru - USP |
dc.source.none.fl_str_mv |
Journal of Applied Oral Science v.28 2020 reponame:Journal of applied oral science (Online) instname:Universidade de São Paulo (USP) instacron:USP |
instname_str |
Universidade de São Paulo (USP) |
instacron_str |
USP |
institution |
USP |
reponame_str |
Journal of applied oral science (Online) |
collection |
Journal of applied oral science (Online) |
repository.name.fl_str_mv |
Journal of applied oral science (Online) - Universidade de São Paulo (USP) |
repository.mail.fl_str_mv |
||jaos@usp.br |
_version_ |
1748936440892358656 |