Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio

Autores
VanWinkle, Peyton E.; Lee, Eunjoo; Wynn, Bridge; Nawara, Tomasz J.; Thomas, Holly; Parant, John; Alvarez, Cecilia Ines; Serra, Rosa; Sztul, Elizabeth
Año de publicación
2024
Idioma
inglés
Tipo de recurso
artículo
Estado
versión publicada
Descripción
BackgroundThe gene cAMP-Responsive Element Binding protein 3-like-1 (CREB3L1) has been implicated in bone development in mice, with CREB3L1 knock-out mice exhibiting fragile bones, and in humans, with CREB3L1 mutations linked to osteogenesis imperfecta. However, the mechanism through which Creb3l1 regulates bone development is not fully understood.ResultsTo probe the role of Creb3l1 in organismal physiology, we used CRISPR-Cas9 genome editing to generate a Danio rerio (zebrafish) model of Creb3l1 deficiency. In contrast to mammalian phenotypes, the Creb3l1 deficient fish do not display abnormalities in osteogenesis, except for a decrease in the bifurcation pattern of caudal fin. Both, skeletal morphology and overall bone density appear normal in the mutant fish. However, the regeneration of caudal fin postamputation is significantly affected, with decreased overall regenerate and mineralized bone area. Moreover, the mutant fish exhibit a severe patterning defect during regeneration, with a significant decrease in bifurcation complexity of the fin rays and distalization of the bifurcation sites. Analysis of genes implicated in bone development showed aberrant patterning of shha and ptch2 in Creb3l1 deficient fish, linking Creb3l1 with Sonic Hedgehog signaling during fin regeneration.ConclusionsOur results uncover a novel role for Creb3l1 in regulating tissue growth and patterning during regeneration.
Fil: VanWinkle, Peyton E.. University of Alabama at Birmingahm; Estados Unidos
Fil: Lee, Eunjoo. University of Alabama at Birmingahm; Estados Unidos
Fil: Wynn, Bridge. University of Alabama at Birmingahm; Estados Unidos
Fil: Nawara, Tomasz J.. University of Alabama at Birmingahm; Estados Unidos
Fil: Thomas, Holly. University of Alabama at Birmingahm; Estados Unidos
Fil: Parant, John. University of Alabama at Birmingahm; Estados Unidos
Fil: Alvarez, Cecilia Ines. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro Científico Tecnológico Córdoba. Centro de Investigaciones en Bioquímica Clínica e Inmunología; Argentina
Fil: Serra, Rosa. University of Alabama at Birmingahm; Estados Unidos
Fil: Sztul, Elizabeth. University of Alabama at Birmingahm; Estados Unidos
Materia
CREB3L1
Nivel de accesibilidad
acceso abierto
Condiciones de uso
https://creativecommons.org/licenses/by-nc/2.5/ar/
Repositorio
CONICET Digital (CONICET)
Institución
Consejo Nacional de Investigaciones Científicas y Técnicas
OAI Identificador
oai:ri.conicet.gov.ar:11336/268558
Acceder
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oai_identifier_str oai:ri.conicet.gov.ar:11336/268558
network_acronym_str CONICETDig
repository_id_str 3498
network_name_str CONICET Digital (CONICET)
spelling Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerioVanWinkle, Peyton E.Lee, EunjooWynn, BridgeNawara, Tomasz J.Thomas, HollyParant, JohnAlvarez, Cecilia InesSerra, RosaSztul, ElizabethCREB3L1https://purl.org/becyt/ford/1.6https://purl.org/becyt/ford/1BackgroundThe gene cAMP-Responsive Element Binding protein 3-like-1 (CREB3L1) has been implicated in bone development in mice, with CREB3L1 knock-out mice exhibiting fragile bones, and in humans, with CREB3L1 mutations linked to osteogenesis imperfecta. However, the mechanism through which Creb3l1 regulates bone development is not fully understood.ResultsTo probe the role of Creb3l1 in organismal physiology, we used CRISPR-Cas9 genome editing to generate a Danio rerio (zebrafish) model of Creb3l1 deficiency. In contrast to mammalian phenotypes, the Creb3l1 deficient fish do not display abnormalities in osteogenesis, except for a decrease in the bifurcation pattern of caudal fin. Both, skeletal morphology and overall bone density appear normal in the mutant fish. However, the regeneration of caudal fin postamputation is significantly affected, with decreased overall regenerate and mineralized bone area. Moreover, the mutant fish exhibit a severe patterning defect during regeneration, with a significant decrease in bifurcation complexity of the fin rays and distalization of the bifurcation sites. Analysis of genes implicated in bone development showed aberrant patterning of shha and ptch2 in Creb3l1 deficient fish, linking Creb3l1 with Sonic Hedgehog signaling during fin regeneration.ConclusionsOur results uncover a novel role for Creb3l1 in regulating tissue growth and patterning during regeneration.Fil: VanWinkle, Peyton E.. University of Alabama at Birmingahm; Estados UnidosFil: Lee, Eunjoo. University of Alabama at Birmingahm; Estados UnidosFil: Wynn, Bridge. University of Alabama at Birmingahm; Estados UnidosFil: Nawara, Tomasz J.. University of Alabama at Birmingahm; Estados UnidosFil: Thomas, Holly. University of Alabama at Birmingahm; Estados UnidosFil: Parant, John. University of Alabama at Birmingahm; Estados UnidosFil: Alvarez, Cecilia Ines. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro Científico Tecnológico Córdoba. Centro de Investigaciones en Bioquímica Clínica e Inmunología; ArgentinaFil: Serra, Rosa. University of Alabama at Birmingahm; Estados UnidosFil: Sztul, Elizabeth. University of Alabama at Birmingahm; Estados UnidosWiley-liss, div John Wiley & Sons Inc.2024-07info:eu-repo/semantics/articleinfo:eu-repo/semantics/publishedVersionhttp://purl.org/coar/resource_type/c_6501info:ar-repo/semantics/articuloapplication/pdfapplication/pdfhttp://hdl.handle.net/11336/268558VanWinkle, Peyton E.; Lee, Eunjoo; Wynn, Bridge; Nawara, Tomasz J.; Thomas, Holly; et al.; Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio; Wiley-liss, div John Wiley & Sons Inc.; Developmental Dynamics; 253; 12; 7-2024; 1106-11291058-8388CONICET DigitalCONICETenginfo:eu-repo/semantics/altIdentifier/url/https://anatomypubs.onlinelibrary.wiley.com/doi/10.1002/dvdy.726info:eu-repo/semantics/altIdentifier/doi/10.1002/dvdy.726info:eu-repo/semantics/openAccesshttps://creativecommons.org/licenses/by-nc/2.5/ar/reponame:CONICET Digital (CONICET)instname:Consejo Nacional de Investigaciones Científicas y Técnicas2025-09-03T09:56:05Zoai:ri.conicet.gov.ar:11336/268558instacron:CONICETInstitucionalhttp://ri.conicet.gov.ar/Organismo científico-tecnológicoNo correspondehttp://ri.conicet.gov.ar/oai/requestdasensio@conicet.gov.ar; lcarlino@conicet.gov.arArgentinaNo correspondeNo correspondeNo correspondeopendoar:34982025-09-03 09:56:06.299CONICET Digital (CONICET) - Consejo Nacional de Investigaciones Científicas y Técnicasfalse
dc.title.none.fl_str_mv Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio
title Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio
spellingShingle Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio
VanWinkle, Peyton E.
CREB3L1
title_short Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio
title_full Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio
title_fullStr Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio
title_full_unstemmed Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio
title_sort Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio
dc.creator.none.fl_str_mv VanWinkle, Peyton E.
Lee, Eunjoo
Wynn, Bridge
Nawara, Tomasz J.
Thomas, Holly
Parant, John
Alvarez, Cecilia Ines
Serra, Rosa
Sztul, Elizabeth
author VanWinkle, Peyton E.
author_facet VanWinkle, Peyton E.
Lee, Eunjoo
Wynn, Bridge
Nawara, Tomasz J.
Thomas, Holly
Parant, John
Alvarez, Cecilia Ines
Serra, Rosa
Sztul, Elizabeth
author_role author
author2 Lee, Eunjoo
Wynn, Bridge
Nawara, Tomasz J.
Thomas, Holly
Parant, John
Alvarez, Cecilia Ines
Serra, Rosa
Sztul, Elizabeth
author2_role author
author
author
author
author
author
author
author
dc.subject.none.fl_str_mv CREB3L1
topic CREB3L1
purl_subject.fl_str_mv https://purl.org/becyt/ford/1.6
https://purl.org/becyt/ford/1
dc.description.none.fl_txt_mv BackgroundThe gene cAMP-Responsive Element Binding protein 3-like-1 (CREB3L1) has been implicated in bone development in mice, with CREB3L1 knock-out mice exhibiting fragile bones, and in humans, with CREB3L1 mutations linked to osteogenesis imperfecta. However, the mechanism through which Creb3l1 regulates bone development is not fully understood.ResultsTo probe the role of Creb3l1 in organismal physiology, we used CRISPR-Cas9 genome editing to generate a Danio rerio (zebrafish) model of Creb3l1 deficiency. In contrast to mammalian phenotypes, the Creb3l1 deficient fish do not display abnormalities in osteogenesis, except for a decrease in the bifurcation pattern of caudal fin. Both, skeletal morphology and overall bone density appear normal in the mutant fish. However, the regeneration of caudal fin postamputation is significantly affected, with decreased overall regenerate and mineralized bone area. Moreover, the mutant fish exhibit a severe patterning defect during regeneration, with a significant decrease in bifurcation complexity of the fin rays and distalization of the bifurcation sites. Analysis of genes implicated in bone development showed aberrant patterning of shha and ptch2 in Creb3l1 deficient fish, linking Creb3l1 with Sonic Hedgehog signaling during fin regeneration.ConclusionsOur results uncover a novel role for Creb3l1 in regulating tissue growth and patterning during regeneration.
Fil: VanWinkle, Peyton E.. University of Alabama at Birmingahm; Estados Unidos
Fil: Lee, Eunjoo. University of Alabama at Birmingahm; Estados Unidos
Fil: Wynn, Bridge. University of Alabama at Birmingahm; Estados Unidos
Fil: Nawara, Tomasz J.. University of Alabama at Birmingahm; Estados Unidos
Fil: Thomas, Holly. University of Alabama at Birmingahm; Estados Unidos
Fil: Parant, John. University of Alabama at Birmingahm; Estados Unidos
Fil: Alvarez, Cecilia Ines. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro Científico Tecnológico Córdoba. Centro de Investigaciones en Bioquímica Clínica e Inmunología; Argentina
Fil: Serra, Rosa. University of Alabama at Birmingahm; Estados Unidos
Fil: Sztul, Elizabeth. University of Alabama at Birmingahm; Estados Unidos
description BackgroundThe gene cAMP-Responsive Element Binding protein 3-like-1 (CREB3L1) has been implicated in bone development in mice, with CREB3L1 knock-out mice exhibiting fragile bones, and in humans, with CREB3L1 mutations linked to osteogenesis imperfecta. However, the mechanism through which Creb3l1 regulates bone development is not fully understood.ResultsTo probe the role of Creb3l1 in organismal physiology, we used CRISPR-Cas9 genome editing to generate a Danio rerio (zebrafish) model of Creb3l1 deficiency. In contrast to mammalian phenotypes, the Creb3l1 deficient fish do not display abnormalities in osteogenesis, except for a decrease in the bifurcation pattern of caudal fin. Both, skeletal morphology and overall bone density appear normal in the mutant fish. However, the regeneration of caudal fin postamputation is significantly affected, with decreased overall regenerate and mineralized bone area. Moreover, the mutant fish exhibit a severe patterning defect during regeneration, with a significant decrease in bifurcation complexity of the fin rays and distalization of the bifurcation sites. Analysis of genes implicated in bone development showed aberrant patterning of shha and ptch2 in Creb3l1 deficient fish, linking Creb3l1 with Sonic Hedgehog signaling during fin regeneration.ConclusionsOur results uncover a novel role for Creb3l1 in regulating tissue growth and patterning during regeneration.
publishDate 2024
dc.date.none.fl_str_mv 2024-07
dc.type.none.fl_str_mv info:eu-repo/semantics/article
info:eu-repo/semantics/publishedVersion
http://purl.org/coar/resource_type/c_6501
info:ar-repo/semantics/articulo
format article
status_str publishedVersion
dc.identifier.none.fl_str_mv http://hdl.handle.net/11336/268558
VanWinkle, Peyton E.; Lee, Eunjoo; Wynn, Bridge; Nawara, Tomasz J.; Thomas, Holly; et al.; Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio; Wiley-liss, div John Wiley & Sons Inc.; Developmental Dynamics; 253; 12; 7-2024; 1106-1129
1058-8388
CONICET Digital
CONICET
url http://hdl.handle.net/11336/268558
identifier_str_mv VanWinkle, Peyton E.; Lee, Eunjoo; Wynn, Bridge; Nawara, Tomasz J.; Thomas, Holly; et al.; Disruption of the creb3l1 gene causes defects in caudal fin regeneration and patterning in zebrafish Danio rerio; Wiley-liss, div John Wiley & Sons Inc.; Developmental Dynamics; 253; 12; 7-2024; 1106-1129
1058-8388
CONICET Digital
CONICET
dc.language.none.fl_str_mv eng
language eng
dc.relation.none.fl_str_mv info:eu-repo/semantics/altIdentifier/url/https://anatomypubs.onlinelibrary.wiley.com/doi/10.1002/dvdy.726
info:eu-repo/semantics/altIdentifier/doi/10.1002/dvdy.726
dc.rights.none.fl_str_mv info:eu-repo/semantics/openAccess
https://creativecommons.org/licenses/by-nc/2.5/ar/
eu_rights_str_mv openAccess
rights_invalid_str_mv https://creativecommons.org/licenses/by-nc/2.5/ar/
dc.format.none.fl_str_mv application/pdf
application/pdf
dc.publisher.none.fl_str_mv Wiley-liss, div John Wiley & Sons Inc.
publisher.none.fl_str_mv Wiley-liss, div John Wiley & Sons Inc.
dc.source.none.fl_str_mv reponame:CONICET Digital (CONICET)
instname:Consejo Nacional de Investigaciones Científicas y Técnicas
reponame_str CONICET Digital (CONICET)
collection CONICET Digital (CONICET)
instname_str Consejo Nacional de Investigaciones Científicas y Técnicas
repository.name.fl_str_mv CONICET Digital (CONICET) - Consejo Nacional de Investigaciones Científicas y Técnicas
repository.mail.fl_str_mv dasensio@conicet.gov.ar; lcarlino@conicet.gov.ar
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score 13.13397

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