ArticlePloS one2013
DiGeorge syndrome gene tbx1 functions through wnt11r to regulate heart looping and differentiation.
Article in PloS one, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers.
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Who cites it
23 citing papers in PubMed, 51 citations in OpenAlex.
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- Wnt9 directs zebrafish heart tube assembly via a combination of canonical and non-canonical pathway signaling.Development (Cambridge, England) · 2023Article
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- From Stripes to a Beating Heart: Early Cardiac Development in Zebrafish.Journal of cardiovascular development and disease · 2021Review
- Exogenous WNT5A and WNT11 proteins rescue CITED2 dysfunction in mouse embryonic stem cells and zebrafish morphants.Cell death & disease · 2019Article
- The Wnt inhibitor Dkk1 is required for maintaining the normal cardiac differentiation program in Xenopus laevis.Developmental biology · 2019Article
- Differential expression of circulating miRNAs as a novel tool to assess BAG3-associated familial dilated cardiomyopathy.Bioscience reports · 2019Article
- Transcriptional regulation of cell shape during organ morphogenesis.The Journal of cell biology · 2018Review
- Continuous addition of progenitors forms the cardiac ventricle in zebrafish.Nature communications · 2018Article
- Distinctive Roles of Canonical and Noncanonical Wnt Signaling in Human Embryonic Cardiomyocyte Development.Stem cell reports · 2016Article
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- Zebrafish Craniofacial Development: A Window into Early Patterning.Current topics in developmental biology · 2015Review
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2 authors at 2 institutions in 1 country.
Funding
Abstract
DiGeorge syndrome (DGS) is the most common microdeletion syndrome, and is characterized by congenital cardiac, craniofacial and immune system abnormalities. The cardiac defects in DGS patients include conotruncal and ventricular septal defects. Although the etiology of DGS is critically regulated by TBX1 gene, the molecular pathways underpinning TBX1's role in heart development are not fully understood. In this study, we characterized heart defects and downstream signaling in the zebrafish tbx1(-/-) mutant, which has craniofacial and immune defects similar to DGS patients. We show that tbx1(-/-) mutants have defective heart looping, morphology and function. Defective heart looping is accompanied by failure of cardiomyocytes to differentiate normally and failure to change shape from isotropic to anisotropic morphology in the outer curvatures of the heart. This is the first demonstration of tbx1's role in regulating heart looping, cardiomyocyte shape and differentiation, and may explain how Tbx1 regulates conotruncal development in humans. Next we elucidated tbx1's molecular signaling pathway guided by the cardiac phenotype of tbx1(-/-) mutants. We show for the first time that wnt11r (wnt11 related), a member of the non-canonical Wnt pathway, and its downstream effector gene alcama (activated leukocyte cell adhesion molecule a) regulate heart looping and differentiation similarly to tbx1. Expression of both wnt11r and alcama are downregulated in tbx1(-/-) mutants. In addition, both wnt11r (-/-) mutants and alcama morphants have heart looping and differentiation defects similar to tbx1(-/-) mutants. Strikingly, heart looping and differentiation in tbx1(-/-) mutants can be partially rescued by ectopic expression of wnt11r or alcama, supporting a model whereby heart looping and differentiation are regulated by tbx1 in a linear pathway through wnt11r and alcama. This is the first study linking tbx1 and non-canonical Wnt signaling and extends our understanding of DGS and heart development.
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