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Mutations in Bcl9 and Pygo genes cause congenital heart defects by tissue-specific perturbation of Wnt/?-catenin signaling.

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

SUBMITTER: Cantu C 

PROVIDER: S-EPMC6217730 | biostudies-literature | 2018 Nov

REPOSITORIES: biostudies-literature

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Bcl9 and Pygopus (Pygo) are obligate Wnt/β-catenin cofactors in <i>Drosophila</i>, yet their contribution to Wnt signaling during vertebrate development remains unresolved. Combining zebrafish and mouse genetics, we document a conserved, β-catenin-associated function for BCL9 and Pygo proteins during vertebrate heart development. Disrupting the β-catenin-BCL9-Pygo complex results in a broadly maintained canonical Wnt response yet perturbs heart development and proper expression of key cardiac re  ...[more]

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