ArticleDevelopmental biology2008
An absence of Twist1 results in aberrant cardiac neural crest morphogenesis.
Article in Developmental biology, 2008. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 52 papers.
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Who cites it
52 citing papers in PubMed, 70 citations in OpenAlex.
- Generation and Characterization of Twist1 Acetyl-Mimic and Acetyl-Deficient Mouse Models.Genesis (New York, N.Y. : 2000) · 2026Article
- Periostin: biology and function in cancer.Cancer cell international · 2022Review
- Single Cell Sequencing Reveals Mechanisms of Persistent Truncus Arteriosus Formation afterGenes · 2022Article
- TheAIMS neuroscience · 2022Review
- TWIST1 transcriptionally upregulates complement 3 in glomerular mesangial cells from spontaneously hypertensive rats.Hypertension research : official journal of the Japanese Society of Hypertension · 2022Article
- The transcription factor Sox7 modulates endocardiac cushion formation contributed to atrioventricular septal defect through Wnt4/Bmp2 signaling.Cell death & disease · 2021Article
- TWIST1 and chromatin regulatory proteins interact to guide neural crest cell differentiation.eLife · 2021Article
- The heart of the neural crest: cardiac neural crest cells in development and regeneration.Development (Cambridge, England) · 2020Review
- Mis-Expression of a Cranial Neural Crest Cell-Specific Gene Program in Cardiac Neural Crest Cells Modulates HAND Factor Expression, Causing Cardiac Outflow Tract Phenotypes.Journal of cardiovascular development and disease · 2020Article
- Evolution and development of the fish jaw skeleton.Wiley interdisciplinary reviews. Developmental biology · 2019Review
- Hand Factors in Cardiac Development.Anatomical record (Hoboken, N.J. : 2007) · 2019Review
- MAPK and PI3K signaling: At the crossroads of neural crest development.Developmental biology · 2018Review
- Congenital heart disease and aortic arch variants associated with mutation in PHOX2B.Genetics in medicine : official journal of the American College of Medical Genetics · 2018Article
- From Neural Crest Development to Cancer and Vice Versa: How p75Frontiers in molecular neuroscience · 2018Review
- The HAND1 frameshift A126FS mutation does not cause hypoplastic left heart syndrome in mice.Cardiovascular research · 2017Article
- Extraocular Muscle Repair and Regeneration.Current ophthalmology reports · 2017Article
- Congenital diaphragmatic hernias: from genes to mechanisms to therapies.Disease models & mechanisms · 2017Review
- Defective Hand1 phosphoregulation uncovers essential roles for Hand1 in limb morphogenesis.Development (Cambridge, England) · 2017Article
- Differential expression pattern of Twist1 in mouse preimplantation embryos suggests its multiple roles during early development.Journal of assisted reproduction and genetics · 2016Article
- Exclusion of Dlx5/6 expression from the distal-most mandibular arches enables BMP-mediated specification of the distal cap.Proceedings of the National Academy of Sciences of the United States of America · 2016Article
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Authors and funding
6 authors at 1 institution in 1 country.
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Abstract
The basic helix-loop-helix transcription factor Twist1 plays an essential role in mesenchymal cell populations during embryonic development and in pathological disease. Remodeling of the cardiac outflow tract (OFT) into the functionally separate aortic arch and pulmonary trunk is dependent upon the dynamic, coordinated contribution of multiple mesenchymal cell populations. Here, we report that Twist1(-/-) mice exhibit OFTs that contain amorphic cellular nodules within their OFT endocardial cushions. The nodular mesenchyme expresses the related bHLH factors Hand1 and Hand2, but reduced levels of the normal cushion marker Periostin. Lineage mapping confirms that nodule cells are exclusively of cardiac neural crest origin (cNCC), and are not ectopic cardiomyocytes or smooth muscle cells. These studies also reveal a delay in cNCC colonization of the OFT cushions. Furthermore, these mapping studies uncover nodules in the pharyngeal arches, and identify Twist1(-/-) neural crest cell defects within the dorsal neural tube, which exhibits an expanded domain of Wnt1-Cre-lineage marked cells. Together, these data support a model where Twist1 is required both for proper cNCC delamination, and for emigration from the dorsal neural tube and along cNCC migration pathways. Within the Twist1(-/-) neural crest cell populations that do emigrate to the OFT, a Hand-expressing subpopulation displays defective maturation, tracking, and, presumably, cell-cell adhesion, further compromising cNCC morphogenesis.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.