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Mutations in Bcl9 and Pygo genes cause congenital heart defects by tissue-specific perturbation of Wnt/ß-catenin signaling.
Cantù, Claudio; Felker, Anastasia; Zimmerli, Dario; Prummel, Karin D; Cabello, Elena M; Chiavacci, Elena; Méndez-Acevedo, Kevin M; Kirchgeorg, Lucia; Burger, Sibylle; Ripoll, Jorge; Valenta, Tomas; Hausmann, George; Vilain, Nathalie; Aguet, Michel; Burger, Alexa; Panáková, Daniela; Basler, Konrad; Mosimann, Christian.
  • Cantù C; Institute of Molecular Life Sciences, University of Zürich, 8057 Zürich, Switzerland.
  • Felker A; Institute of Molecular Life Sciences, University of Zürich, 8057 Zürich, Switzerland.
  • Zimmerli D; Institute of Molecular Life Sciences, University of Zürich, 8057 Zürich, Switzerland.
  • Prummel KD; Institute of Molecular Life Sciences, University of Zürich, 8057 Zürich, Switzerland.
  • Cabello EM; Institute of Molecular Life Sciences, University of Zürich, 8057 Zürich, Switzerland.
  • Chiavacci E; Institute of Molecular Life Sciences, University of Zürich, 8057 Zürich, Switzerland.
  • Méndez-Acevedo KM; Electrochemical Signaling in Development and Disease, Max Delbrück Center for Molecular Medicine in the Helmholtz Association, 13125 Berlin-Buch, Germany.
  • Kirchgeorg L; Institute of Molecular Life Sciences, University of Zürich, 8057 Zürich, Switzerland.
  • Burger S; Institute of Molecular Life Sciences, University of Zürich, 8057 Zürich, Switzerland.
  • Ripoll J; Department of Bioengineering and Aerospace Engineering, Universidad Carlos III de Madrid, 28911 Madrid, Spain.
  • Valenta T; Institute of Molecular Life Sciences, University of Zürich, 8057 Zürich, Switzerland.
  • Hausmann G; Institute of Molecular Life Sciences, University of Zürich, 8057 Zürich, Switzerland.
  • Vilain N; Swiss Institute for Experimental Cancer Research (ISREC), École Polytechnique Fédérale de Lausanne (EPFL), School of Life Sciences, 1015 Lausanne, Switzerland.
  • Aguet M; Swiss Institute for Experimental Cancer Research (ISREC), École Polytechnique Fédérale de Lausanne (EPFL), School of Life Sciences, 1015 Lausanne, Switzerland.
  • Burger A; Institute of Molecular Life Sciences, University of Zürich, 8057 Zürich, Switzerland.
  • Panáková D; Electrochemical Signaling in Development and Disease, Max Delbrück Center for Molecular Medicine in the Helmholtz Association, 13125 Berlin-Buch, Germany.
  • Basler K; Deutsches Zentrum für Herz-Kreislauf-Forschung (DZHK), partner site Berlin, 10115 Berlin, Germany.
  • Mosimann C; Institute of Molecular Life Sciences, University of Zürich, 8057 Zürich, Switzerland.
Genes Dev ; 32(21-22): 1443-1458, 2018 11 01.
Article en En | MEDLINE | ID: mdl-30366904
Bcl9 and Pygopus (Pygo) are obligate Wnt/ß-catenin cofactors in Drosophila, yet their contribution to Wnt signaling during vertebrate development remains unresolved. Combining zebrafish and mouse genetics, we document a conserved, ß-catenin-associated function for BCL9 and Pygo proteins during vertebrate heart development. Disrupting the ß-catenin-BCL9-Pygo complex results in a broadly maintained canonical Wnt response yet perturbs heart development and proper expression of key cardiac regulators. Our work highlights BCL9 and Pygo as selective ß-catenin cofactors in a subset of canonical Wnt responses during vertebrate development. Moreover, our results implicate alterations in BCL9 and BCL9L in human congenital heart defects.
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Texto completo: 1 Banco de datos: MEDLINE Asunto principal: Factores de Transcripción / Proteínas de Pez Cebra / Péptidos y Proteínas de Señalización Intracelular / Vía de Señalización Wnt / Cardiopatías Congénitas Límite: Animals Idioma: En Año: 2018 Tipo del documento: Article
Texto completo
Imprimir
XML
PubMed Links
Search on Google

Texto completo: 1 Banco de datos: MEDLINE Asunto principal: Factores de Transcripción / Proteínas de Pez Cebra / Péptidos y Proteínas de Señalización Intracelular / Vía de Señalización Wnt / Cardiopatías Congénitas Límite: Animals Idioma: En Año: 2018 Tipo del documento: Article