First Author | Cantù C | Year | 2018 |
Journal | Genes Dev | Volume | 32 |
Issue | 21-22 | Pages | 1443-1458 |
PubMed ID | 30366904 | Mgi Jnum | J:272561 |
Mgi Id | MGI:6284056 | Doi | 10.1101/gad.315531.118 |
Citation | Cantu C, et al. (2018) Mutations in Bcl9 and Pygo genes cause congenital heart defects by tissue-specific perturbation of Wnt/beta-catenin signaling. Genes Dev 32(21-22):1443-1458 |
abstractText | 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. |