| First Author | Johnson AL | Year | 2020 |
| Journal | J Cardiovasc Dev Dis | Volume | 7 |
| Issue | 3 | PubMed ID | 32717817 |
| Mgi Jnum | J:320915 | Mgi Id | MGI:6883533 |
| Doi | 10.3390/jcdd7030027 | Citation | Johnson AL, et al. (2020) Early Embryonic Expression of AP-2alpha Is Critical for Cardiovascular Development. J Cardiovasc Dev Dis 7(3):27 |
| abstractText | Congenital cardiovascular malformation is a common birth defect incorporating abnormalities of the outflow tract and aortic arch arteries, and mice deficient in the transcription factor AP-2alpha (Tcfap2a) present with complex defects affecting these structures. AP-2alpha is expressed in the pharyngeal surface ectoderm and neural crest at mid-embryogenesis in the mouse, but the precise tissue compartment in which AP-2alpha is required for cardiovascular development has not been identified. In this study we describe the fully penetrant AP-2alpha deficient cardiovascular phenotype on a C57Bl/6J genetic background and show that this is associated with increased apoptosis in the pharyngeal ectoderm. Neural crest cell migration into the pharyngeal arches was not affected. Cre-expressing transgenic mice were used in conjunction with an AP-2alpha conditional allele to examine the effect of deleting AP-2alpha from the pharyngeal surface ectoderm and the neural crest, either individually or in combination, as well as the second heart field. This, surprisingly, was unable to fully recapitulate the global AP-2alpha deficient cardiovascular phenotype. The outflow tract and arch artery phenotype was, however, recapitulated through early embryonic Cre-mediated recombination. These findings indicate that AP-2alpha has a complex influence on cardiovascular development either being required very early in embryogenesis and/or having a redundant function in many tissue layers. |
