ArticleMechanisms of development
Cardiac outflow tract septation failure in Pax3-deficient embryos is due to p53-dependent regulation of migrating cardiac neural crest.
Article in Mechanisms of development. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 30 papers.
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Who cites it
30 citing papers in PubMed, 54 citations in OpenAlex.
- PAX3: A Driver of Normal Development and Disease.Biomolecules · 2026Review
- PAX Genes in Cardiovascular Development.International journal of molecular sciences · 2022Review
- Perioperative Care of a Patient With Waardenburg Syndrome.Journal of medical cases · 2021Article
- Prevention of Teratogenesis in Pregnancies of Obese Rats by Vitamin E Supplementation.Antioxidants (Basel, Switzerland) · 2021Article
- The role of glucose in physiological and pathological heart formation.Developmental biology · 2021Review
- Mutation in Eftud2 causes craniofacial defects in mice via mis-splicing of Mdm2 and increased P53.Human molecular genetics · 2021Article
- Mechanisms of Congenital Malformations in Pregnancies with Pre-existing Diabetes.Current diabetes reports · 2020Review
- The Spatiotemporal Pattern and Intensity of p53 Activation Dictates Phenotypic Diversity in p53-Driven Developmental Syndromes.Developmental cell · 2019Article
- The role of p53 in developmental syndromes.Journal of molecular cell biology · 2019Review
- Autophagy-dependent crosstalk between GILT and PAX-3 influences radiation sensitivity of human melanoma cells.Journal of cellular biochemistry · 2018Article
- Pdgfrα functions in endothelial-derived cells to regulate neural crest cells and the development of the great arteries.Disease models & mechanisms · 2017Article
- microRNA expression profiling and functional annotation analysis of their targets modulated by oxidative stress during embryonic heart development in diabetic mice.Reproductive toxicology (Elmsford, N.Y.) · 2016Article
- The status of diabetic embryopathy.Upsala journal of medical sciences · 2016Review
- Deregulated Cardiac Specific MicroRNAs in Postnatal Heart Growth.BioMed research international · 2016Article
- Hyperglycemia impairs left-right axis formation and thereby disturbs heart morphogenesis in mouse embryos.Proceedings of the National Academy of Sciences of the United States of America · 2015Article
- TGFβ and Wnt in cardiac outflow tract defects in offspring of diabetic pregnancies.Birth defects research. Part B, Developmental and reproductive toxicology · 2014Article
- Article
- Guilty as CHARGED: p53's expanding role in disease.Cell cycle (Georgetown, Tex.) · 2014Article
- Familial dysautonomia model reveals Ikbkap deletion causes apoptosis of Pax3+ progenitors and peripheral neurons.Proceedings of the National Academy of Sciences of the United States of America · 2013Article
- Tcof1 acts as a modifier of Pax3 during enteric nervous system development and in the pathogenesis of colonic aganglionosis.Human molecular genetics · 2013Article
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Authors and funding
6 authors at 4 institutions in 2 countries.
Funding
Abstract
During neural tube closure, Pax3 is required to inhibit p53-dependent apoptosis. Pax3 is also required for migration of cardiac neural crest (CNC) from the neural tube to the heart and septation of the primitive single cardiac outflow tract into the aorta and pulmonary arteries. Whether Pax3 is required for CNC migration and outflow tract septation by inhibiting p53-dependent apoptosis is not known. In this study, mouse strains carrying reporters linked to Pax3 alleles were used to map the fate of CNC cells in embryos which were either Pax3-sufficient (expressing one or two functional Pax3 alleles) or Pax3-deficient (expressing two null Pax3 alleles), and in which p53 had been inactivated or not. Migrating CNC cells were observed in both Pax3-sufficient and -deficient embryos, but CNC cells were sparse and disorganized in Pax3-deficient embryos as migration progressed. The defective migration was associated with increased cell death. Suppression of p53, either by null mutation of the p53 gene, or administration of a p53 inhibitor, pifithrin-alpha, prevented the defective CNC migration and apoptosis in Pax3-deficient embryos, and also restored proper development of cardiac outflow tracts. These results indicate that Pax3 is required for cardiac outflow tract septation because it blocks p53-dependent processes during CNC migration.
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