ArticleMechanisms of development
Williams Syndrome Transcription Factor is critical for neural crest cell function in Xenopus laevis.
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 27 papers.
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Who cites it
27 citing papers in PubMed, 52 citations in OpenAlex.
- WSTF deficiency reprograms regulatory networks by linking locus-specific chromatin remodeling to altered isoform expression and misdirected signaling.Nucleic acids research · 2026Article
- Review
- The chromatin regulator Ankrd11 controls cardiac neural crest cell-mediated outflow tract remodeling and heart function.Nature communications · 2024Article
- Epigenetic regulation of craniofacial development and disease.Birth defects research · 2024Review
- Loss of Baz1b in mice causes perinatal lethality, growth failure, and variable multi-system outcomes.Developmental biology · 2024Article
- Novel ophthalmic findings and deep phenotyping in Williams-Beuren syndrome.The British journal of ophthalmology · 2023Article
- Adult William's Syndrome: The Cause of an Unusual Vasculopathy and Biliary Abnormalities.Cureus · 2023Article
- Article
- Evidence supporting the oncogenic role of BAZ1B in colorectal cancer.American journal of cancer research · 2022Article
- BAZ1B the Protean Protein.Genes · 2021Review
- Antagonising Chromatin Remodelling Activities in the Regulation of Mammalian Ribosomal Transcription.Genes · 2021Review
- Williams syndrome.Nature reviews. Disease primers · 2021Review
- Epigenetic Regulation of Cardiac Neural Crest Cells.Frontiers in cell and developmental biology · 2021Review
- Birdsong as a window into language origins and evolutionary neuroscience.Philosophical transactions of the Royal Society of London. Series B, Biological sciences · 2020Review
- Dosage analysis of the 7q11.23 Williams region identifiesScience advances · 2019Article
- Epigenetic Mechanisms in Hirschsprung Disease.International journal of molecular sciences · 2019Review
- Neural crest contributions to the ear: Implications for congenital hearing disorders.Hearing research · 2019Review
- Williams Syndrome, Human Self-Domestication, and Language Evolution.Frontiers in psychology · 2019Article
- Comparison of village dog and wolf genomes highlights the role of the neural crest in dog domestication.BMC biology · 2018Article
- Review
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Authors and funding
9 authors at 2 institutions in 1 country.
Funding
Abstract
Williams Syndrome Transcription Factor (WSTF) is one of ∼25 haplodeficient genes in patients with the complex developmental disorder Williams Syndrome (WS). WS results in visual/spatial processing defects, cognitive impairment, unique behavioral phenotypes, characteristic "elfin" facial features, low muscle tone and heart defects. WSTF exists in several chromatin remodeling complexes and has roles in transcription, replication, and repair. Chromatin remodeling is essential during embryogenesis, but WSTF's role in vertebrate development is poorly characterized. To investigate the developmental role of WSTF, we knocked down WSTF in Xenopus laevis embryos using a morpholino that targets WSTF mRNA. BMP4 shows markedly increased and spatially aberrant expression in WSTF-deficient embryos, while SHH, MRF4, PAX2, EPHA4 and SOX2 expression are severely reduced, coupled with defects in a number of developing embryonic structures and organs. WSTF-deficient embryos display defects in anterior neural development. Induction of the neural crest, measured by expression of the neural crest-specific genes SNAIL and SLUG, is unaffected by WSTF depletion. However, at subsequent stages WSTF knockdown results in a severe defect in neural crest migration and/or maintenance. Consistent with a maintenance defect, WSTF knockdowns display a specific pattern of increased apoptosis at the tailbud stage in regions corresponding to the path of cranial neural crest migration. Our work is the first to describe a role for WSTF in proper neural crest function, and suggests that neural crest defects resulting from WSTF haploinsufficiency may be a major contributor to the pathoembryology of WS.
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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.