Rathke’s cleft-like cysts arise from Isl1 deletion in murine pituitary progenitors

Autores
Brinkmeier, Michelle L.; Bando, Hironori; Camarano, Adriana Carla; Fujio, Shingo; Yoshimoto, Koji; Silva Junqueira de Souza, Flávio; Camper, Sally
Año de publicación
2020
Idioma
inglés
Tipo de recurso
artículo
Estado
versión publicada
Descripción
The transcription factor ISL1 is expressed in pituitary gland stem cells and the thyrotrope and gonadotrope lineages. Pituitary-specific Isl1 deletion causes hypopituitarism with increased stem cell apoptosis, reduced differentiation of thyrotropes and gonadotropes, and reduced body size. Conditional Isl1 deletion causes development of multiple Rathke’s cleft-like cysts, with 100% penetrance. Foxa1 and Foxj1 are abnormally expressed in the pituitary gland and associated with a ciliogenic gene-expression program in the cysts. We confirmed expression of FOXA1, FOXJ1, and stem cell markers in human Rathke’s cleft cyst tissue, but not craniopharyngiomas, which suggests these transcription factors are useful, pathological markers for diagnosis of Rathke’s cleft cysts. These studies support a model whereby expression of ISL1 in pituitary progenitors drives differentiation into thyrotropes and gonadotropes and without it, activation of FOXA1 and FOXJ1 permits development of an oral epithelial cell fate with mucinous cysts. This pituitary-specific Isl1 mouse knockout sheds light on the etiology of Rathke’s cleft cysts and the role of ISL1 in normal pituitary development.
Fil: Brinkmeier, Michelle L.. University of Michigan; Estados Unidos
Fil: Bando, Hironori. University of Michigan; Estados Unidos
Fil: Camarano, Adriana Carla. Consejo Nacional de Investigaciones Científicas y Técnicas. Oficina de Coordinación Administrativa Ciudad Universitaria. Instituto de Fisiología, Biología Molecular y Neurociencias. Universidad de Buenos Aires. Facultad de Ciencias Exactas y Naturales. Instituto de Fisiología, Biología Molecular y Neurociencias; Argentina
Fil: Fujio, Shingo. Kagoshima University; Japón
Fil: Yoshimoto, Koji. Kagoshima University; Japón
Fil: Silva Junqueira de Souza, Flávio. Consejo Nacional de Investigaciones Científicas y Técnicas. Oficina de Coordinación Administrativa Ciudad Universitaria. Instituto de Fisiología, Biología Molecular y Neurociencias. Universidad de Buenos Aires. Facultad de Ciencias Exactas y Naturales. Instituto de Fisiología, Biología Molecular y Neurociencias; Argentina
Fil: Camper, Sally. University of Michigan; Estados Unidos
Materia
RATHKE'SCLEFT-LIKECYSTS
ISL1
PITUITARY
PROGENITORS
Nivel de accesibilidad
acceso abierto
Condiciones de uso
https://creativecommons.org/licenses/by-nc-sa/2.5/ar/
Repositorio
CONICET Digital (CONICET)
Institución
Consejo Nacional de Investigaciones Científicas y Técnicas
OAI Identificador
oai:ri.conicet.gov.ar:11336/142344
Acceder
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oai_identifier_str oai:ri.conicet.gov.ar:11336/142344
network_acronym_str CONICETDig
repository_id_str 3498
network_name_str CONICET Digital (CONICET)
spelling Rathke’s cleft-like cysts arise from Isl1 deletion in murine pituitary progenitorsBrinkmeier, Michelle L.Bando, HironoriCamarano, Adriana CarlaFujio, ShingoYoshimoto, KojiSilva Junqueira de Souza, FlávioCamper, SallyRATHKE'SCLEFT-LIKECYSTSISL1PITUITARYPROGENITORShttps://purl.org/becyt/ford/1.6https://purl.org/becyt/ford/1The transcription factor ISL1 is expressed in pituitary gland stem cells and the thyrotrope and gonadotrope lineages. Pituitary-specific Isl1 deletion causes hypopituitarism with increased stem cell apoptosis, reduced differentiation of thyrotropes and gonadotropes, and reduced body size. Conditional Isl1 deletion causes development of multiple Rathke’s cleft-like cysts, with 100% penetrance. Foxa1 and Foxj1 are abnormally expressed in the pituitary gland and associated with a ciliogenic gene-expression program in the cysts. We confirmed expression of FOXA1, FOXJ1, and stem cell markers in human Rathke’s cleft cyst tissue, but not craniopharyngiomas, which suggests these transcription factors are useful, pathological markers for diagnosis of Rathke’s cleft cysts. These studies support a model whereby expression of ISL1 in pituitary progenitors drives differentiation into thyrotropes and gonadotropes and without it, activation of FOXA1 and FOXJ1 permits development of an oral epithelial cell fate with mucinous cysts. This pituitary-specific Isl1 mouse knockout sheds light on the etiology of Rathke’s cleft cysts and the role of ISL1 in normal pituitary development.Fil: Brinkmeier, Michelle L.. University of Michigan; Estados UnidosFil: Bando, Hironori. University of Michigan; Estados UnidosFil: Camarano, Adriana Carla. Consejo Nacional de Investigaciones Científicas y Técnicas. Oficina de Coordinación Administrativa Ciudad Universitaria. Instituto de Fisiología, Biología Molecular y Neurociencias. Universidad de Buenos Aires. Facultad de Ciencias Exactas y Naturales. Instituto de Fisiología, Biología Molecular y Neurociencias; ArgentinaFil: Fujio, Shingo. Kagoshima University; JapónFil: Yoshimoto, Koji. Kagoshima University; JapónFil: Silva Junqueira de Souza, Flávio. Consejo Nacional de Investigaciones Científicas y Técnicas. Oficina de Coordinación Administrativa Ciudad Universitaria. Instituto de Fisiología, Biología Molecular y Neurociencias. Universidad de Buenos Aires. Facultad de Ciencias Exactas y Naturales. Instituto de Fisiología, Biología Molecular y Neurociencias; ArgentinaFil: Camper, Sally. University of Michigan; Estados UnidosAmerican Society for Clinical Investigation2020-08info:eu-repo/semantics/articleinfo:eu-repo/semantics/publishedVersionhttp://purl.org/coar/resource_type/c_6501info:ar-repo/semantics/articuloapplication/pdfapplication/pdfhttp://hdl.handle.net/11336/142344Brinkmeier, Michelle L.; Bando, Hironori; Camarano, Adriana Carla; Fujio, Shingo; Yoshimoto, Koji; et al.; Rathke’s cleft-like cysts arise from Isl1 deletion in murine pituitary progenitors; American Society for Clinical Investigation; Journal of Clinical Investigation; 140; 8; 8-2020; 4501-45150021-9738CONICET DigitalCONICETenginfo:eu-repo/semantics/altIdentifier/doi/10.1172/JCI136745info:eu-repo/semantics/altIdentifier/url/https://www.jci.org/articles/view/136745info:eu-repo/semantics/openAccesshttps://creativecommons.org/licenses/by-nc-sa/2.5/ar/reponame:CONICET Digital (CONICET)instname:Consejo Nacional de Investigaciones Científicas y Técnicas2025-09-10T13:13:11Zoai:ri.conicet.gov.ar:11336/142344instacron: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:34982025-09-10 13:13:12.01CONICET Digital (CONICET) - Consejo Nacional de Investigaciones Científicas y Técnicasfalse
dc.title.none.fl_str_mv Rathke’s cleft-like cysts arise from Isl1 deletion in murine pituitary progenitors
title Rathke’s cleft-like cysts arise from Isl1 deletion in murine pituitary progenitors
spellingShingle Rathke’s cleft-like cysts arise from Isl1 deletion in murine pituitary progenitors
Brinkmeier, Michelle L.
RATHKE'SCLEFT-LIKECYSTS
ISL1
PITUITARY
PROGENITORS
title_short Rathke’s cleft-like cysts arise from Isl1 deletion in murine pituitary progenitors
title_full Rathke’s cleft-like cysts arise from Isl1 deletion in murine pituitary progenitors
title_fullStr Rathke’s cleft-like cysts arise from Isl1 deletion in murine pituitary progenitors
title_full_unstemmed Rathke’s cleft-like cysts arise from Isl1 deletion in murine pituitary progenitors
title_sort Rathke’s cleft-like cysts arise from Isl1 deletion in murine pituitary progenitors
dc.creator.none.fl_str_mv Brinkmeier, Michelle L.
Bando, Hironori
Camarano, Adriana Carla
Fujio, Shingo
Yoshimoto, Koji
Silva Junqueira de Souza, Flávio
Camper, Sally
author Brinkmeier, Michelle L.
author_facet Brinkmeier, Michelle L.
Bando, Hironori
Camarano, Adriana Carla
Fujio, Shingo
Yoshimoto, Koji
Silva Junqueira de Souza, Flávio
Camper, Sally
author_role author
author2 Bando, Hironori
Camarano, Adriana Carla
Fujio, Shingo
Yoshimoto, Koji
Silva Junqueira de Souza, Flávio
Camper, Sally
author2_role author
author
author
author
author
author
dc.subject.none.fl_str_mv RATHKE'SCLEFT-LIKECYSTS
ISL1
PITUITARY
PROGENITORS
topic RATHKE'SCLEFT-LIKECYSTS
ISL1
PITUITARY
PROGENITORS
purl_subject.fl_str_mv https://purl.org/becyt/ford/1.6
https://purl.org/becyt/ford/1
dc.description.none.fl_txt_mv The transcription factor ISL1 is expressed in pituitary gland stem cells and the thyrotrope and gonadotrope lineages. Pituitary-specific Isl1 deletion causes hypopituitarism with increased stem cell apoptosis, reduced differentiation of thyrotropes and gonadotropes, and reduced body size. Conditional Isl1 deletion causes development of multiple Rathke’s cleft-like cysts, with 100% penetrance. Foxa1 and Foxj1 are abnormally expressed in the pituitary gland and associated with a ciliogenic gene-expression program in the cysts. We confirmed expression of FOXA1, FOXJ1, and stem cell markers in human Rathke’s cleft cyst tissue, but not craniopharyngiomas, which suggests these transcription factors are useful, pathological markers for diagnosis of Rathke’s cleft cysts. These studies support a model whereby expression of ISL1 in pituitary progenitors drives differentiation into thyrotropes and gonadotropes and without it, activation of FOXA1 and FOXJ1 permits development of an oral epithelial cell fate with mucinous cysts. This pituitary-specific Isl1 mouse knockout sheds light on the etiology of Rathke’s cleft cysts and the role of ISL1 in normal pituitary development.
Fil: Brinkmeier, Michelle L.. University of Michigan; Estados Unidos
Fil: Bando, Hironori. University of Michigan; Estados Unidos
Fil: Camarano, Adriana Carla. Consejo Nacional de Investigaciones Científicas y Técnicas. Oficina de Coordinación Administrativa Ciudad Universitaria. Instituto de Fisiología, Biología Molecular y Neurociencias. Universidad de Buenos Aires. Facultad de Ciencias Exactas y Naturales. Instituto de Fisiología, Biología Molecular y Neurociencias; Argentina
Fil: Fujio, Shingo. Kagoshima University; Japón
Fil: Yoshimoto, Koji. Kagoshima University; Japón
Fil: Silva Junqueira de Souza, Flávio. Consejo Nacional de Investigaciones Científicas y Técnicas. Oficina de Coordinación Administrativa Ciudad Universitaria. Instituto de Fisiología, Biología Molecular y Neurociencias. Universidad de Buenos Aires. Facultad de Ciencias Exactas y Naturales. Instituto de Fisiología, Biología Molecular y Neurociencias; Argentina
Fil: Camper, Sally. University of Michigan; Estados Unidos
description The transcription factor ISL1 is expressed in pituitary gland stem cells and the thyrotrope and gonadotrope lineages. Pituitary-specific Isl1 deletion causes hypopituitarism with increased stem cell apoptosis, reduced differentiation of thyrotropes and gonadotropes, and reduced body size. Conditional Isl1 deletion causes development of multiple Rathke’s cleft-like cysts, with 100% penetrance. Foxa1 and Foxj1 are abnormally expressed in the pituitary gland and associated with a ciliogenic gene-expression program in the cysts. We confirmed expression of FOXA1, FOXJ1, and stem cell markers in human Rathke’s cleft cyst tissue, but not craniopharyngiomas, which suggests these transcription factors are useful, pathological markers for diagnosis of Rathke’s cleft cysts. These studies support a model whereby expression of ISL1 in pituitary progenitors drives differentiation into thyrotropes and gonadotropes and without it, activation of FOXA1 and FOXJ1 permits development of an oral epithelial cell fate with mucinous cysts. This pituitary-specific Isl1 mouse knockout sheds light on the etiology of Rathke’s cleft cysts and the role of ISL1 in normal pituitary development.
publishDate 2020
dc.date.none.fl_str_mv 2020-08
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/11336/142344
Brinkmeier, Michelle L.; Bando, Hironori; Camarano, Adriana Carla; Fujio, Shingo; Yoshimoto, Koji; et al.; Rathke’s cleft-like cysts arise from Isl1 deletion in murine pituitary progenitors; American Society for Clinical Investigation; Journal of Clinical Investigation; 140; 8; 8-2020; 4501-4515
0021-9738
CONICET Digital
CONICET
url http://hdl.handle.net/11336/142344
identifier_str_mv Brinkmeier, Michelle L.; Bando, Hironori; Camarano, Adriana Carla; Fujio, Shingo; Yoshimoto, Koji; et al.; Rathke’s cleft-like cysts arise from Isl1 deletion in murine pituitary progenitors; American Society for Clinical Investigation; Journal of Clinical Investigation; 140; 8; 8-2020; 4501-4515
0021-9738
CONICET Digital
CONICET
dc.language.none.fl_str_mv eng
language eng
dc.relation.none.fl_str_mv info:eu-repo/semantics/altIdentifier/doi/10.1172/JCI136745
info:eu-repo/semantics/altIdentifier/url/https://www.jci.org/articles/view/136745
dc.rights.none.fl_str_mv info:eu-repo/semantics/openAccess
https://creativecommons.org/licenses/by-nc-sa/2.5/ar/
eu_rights_str_mv openAccess
rights_invalid_str_mv https://creativecommons.org/licenses/by-nc-sa/2.5/ar/
dc.format.none.fl_str_mv application/pdf
application/pdf
dc.publisher.none.fl_str_mv American Society for Clinical Investigation
publisher.none.fl_str_mv American Society for Clinical Investigation
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
_version_ 1842980694959063040
score 12.993085

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