ArticleDevelopmental dynamics : an official publication of the American Association of Anatomists2019
Nonlinear gene expression-phenotype relationships contribute to variation and clefting in the A/WySn mouse.
Article in Developmental dynamics : an official publication of the American Association of Anatomists, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers.
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Who cites it
11 citing papers in PubMed, 23 citations in OpenAlex.
- Navigating the window: structural bottlenecks in avian craniofacial integration revealed by the ontogenetic patterning of shape variance.bioRxiv : the preprint server for biology · 2026Article
- Article
- Rapid and biased evolution of canalization during adaptive divergence revealed by dominance in gene expression variability during Arctic charr early development.Communications biology · 2023Article
- Adding gene transcripts into genomic prediction improves accuracy and reveals sampling time dependence.G3 (Bethesda, Md.) · 2022Article
- Alterations in DNA Methylation in Orofacial Clefts.International journal of molecular sciences · 2022Review
- Extend mixed models to multilayer neural networks for genomic prediction including intermediate omics data.Genetics · 2022Article
- What's Shape Got to Do With It? Examining the Relationship Between Facial Shape and Orofacial Clefting.Frontiers in genetics · 2022Review
- Editorial: Cellular Mechanisms During Normal and Abnormal Craniofacial Development.Frontiers in cell and developmental biology · 2022Article
- Article
- Review
- Phosphotungstic acid-enhanced microCT: Optimized protocols for embryonic and early postnatal mice.Developmental dynamics : an official publication of the American Association of Anatomists · 2020Article
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Authors and funding
10 authors at 6 institutions in 2 countries.
Funding
Abstract
backgroundCleft lip and palate is one of the most common human birth defects, but the underlying etiology is poorly understood. The A/WySn mouse is a spontaneously occurring model of multigenic clefting in which 20% to 30% of individuals develop an orofacial cleft. Recent work has shown altered methylation at a specific retrotransposon insertion downstream of the Wnt9b locus in clefting animals, which results in decreased Wnt9b expression.
resultsUsing a newly developed protocol that allows us to measure morphology, gene expression, and DNA methylation in the same embryo, we relate gene expression in an individual embryo directly to its three-dimensional morphology for the first time. We find that methylation at the retrotransposon relates to Wnt9b expression and morphology. IAP methylation relates to shape of the nasal process in a manner consistent with clefting. Embryos with low IAP methylation exhibit increased among-individual variance in facial shape.
conclusionsMethylation and gene expression relate nonlinearly to nasal process morphology. Individuals at one end of a continuum of phenotypic states display a clinical phenotype and increased phenotypic variation. Variable penetrance and expressivity in this model is likely determined both by among-individual variation in methylation and changes in phenotypic robustness along the underlying liability distribution for orofacial clefting.
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