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Mutations in Bcl9 and Pygo genes cause congenital heart defects by tissue-specific perturbation of Wnt/beta-catenin signaling
Linköping University, Department of Clinical and Experimental Medicine, Division of Microbiology and Molecular Medicine. Linköping University, Faculty of Medicine and Health Sciences. Univ Zurich, Switzerland.
Univ Zurich, Switzerland.
Univ Zurich, Switzerland.
Univ Zurich, Switzerland.
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2018 (English)In: Genes & Development, ISSN 0890-9369, E-ISSN 1549-5477, Vol. 32, no 21-22, p. 1443-1458Article in journal (Refereed) Published
Abstract [en]

Bcl9 and Pygopus (Pygo) are obligate Wnt/beta-catenin cofactors in Drosophila, yet their contribution to Wnt signaling during vertebrate development remains unresolved. Combining zebrafish and mouse genetics, we document a conserved, beta-catenin-associated function for BCL9 and Pygo proteins during vertebrate heart development. Disrupting the beta-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 beta-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.

Place, publisher, year, edition, pages
COLD SPRING HARBOR LAB PRESS, PUBLICATIONS DEPT , 2018. Vol. 32, no 21-22, p. 1443-1458
Keywords [en]
CRISPR-Cas9; cardiovascular development; congenital heart disease; heart; transcription; Wnt signaling
National Category
Developmental Biology
Identifiers
URN: urn:nbn:se:liu:diva-153382DOI: 10.1101/gad.315531.118ISI: 000451237000008PubMedID: 30366904OAI: oai:DiVA.org:liu-153382DiVA, id: diva2:1271793
Note

Funding Agencies|Swiss National Science Foundation (SNSF) [PP00P3_139093]; SNSF REquip grant [150838]; European Commission [CIG PCIG14-GA-2013-631984]; Canton of Zurich; UZH Foundation for Research in Science and the Humanities; Swiss Heart Foundation; SSNF; Forschungskredit of UZH; Ministry of Economy and Competitiveness (MINECO) [FIS2016-77892-R]

Available from: 2018-12-18 Created: 2018-12-18 Last updated: 2019-03-22

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Cantù, Claudio
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