Dataset Information

Palatal segment contributions to midfacial growth in three inbred mouse strains.

ABSTRACT: Following facial prominence fusion, anterior-posterior (A-P) elongation of the palate is a critical aspect of palatogenesis and integrated midfacial elongation. Reciprocal epithelial-mesenchymal interactions drive secondary palate elongation and periodic signaling center formation within the rugae growth zone (RGZ). However, the relationship between RGZ dynamics and the morphogenetic behavior of underlying palatal bone mesenchymal precursors has remained enigmatic. Our results indicate that cellular activity at the RGZ simultaneously drives rugae formation and elongation of the maxillary bone primordium within the anterior secondary palate, which more than doubles in length prior to palatal shelf fusion. The first formed palatal ruga, found just posterior to the RGZ, represents a consistent morphological boundary between anterior and posterior secondary palate bone precursors, being found at the future maxillary-palatine suture. These results suggest a model where changes in RGZ-driven A-P growth of the anterior secondary palate may produce interspecies and intraspecies differences in facial prognathism and differences in the proportional contribution of palatal segment-associated bones to total palate length. An ontogenetic comparison of three inbred mouse strains indicated that while RGZ-driven growth of the anterior secondary palate is critical for early midfacial outgrowth, subtle strain-specific bony contributions to adult palate length are not present until after this initial palatal growth period. This multifaceted illustration of normal midfacial growth dynamics confirms a one-to-one relationship between palatal segments and upper jaw bones during the earliest stages of palatal growth, which may serve as the basis for evolutionary change in upper jaw morphology. Additionally, identified mouse strain-specific differences in palatal segment elongation provide a useful foundation for understanding the impact of background genetic effects on facial morphogenesis.

SUBMITTER: Welsh IC

PROVIDER: S-EPMC10592893 | biostudies-literature | 2023 Oct

REPOSITORIES: biostudies-literature

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Palatal segment contributions to midfacial anterior-posterior growth.

Welsh Ian C IC Feiler Maria E ME Lipman Danika D Mormile Isabel I Hansen Karissa K Percival Christopher J CJ

bioRxiv : the preprint server for biology 20241113

Anterior-posterior (A-P) elongation of the palate is a critical aspect of integrated midfacial morphogenesis. Reciprocal epithelial-mesenchymal interactions drive secondary palate elongation that is coupled to the periodic formation of signaling centers within the rugae growth zone (RGZ). However, the relationship between RGZ-driven morphogenetic processes, the differentiative dynamics of underlying palatal bone mesenchymal precursors, and the segmental organization of the upper jaw has remained ...[more]

PMID: 37873353

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Project description:Mice are the most widely used animal model to study genotype to phenotype relationships. Inbred mice are genetically identical, which eliminates genetic heterogeneity and makes them particularly useful for genetic studies. Many different strains have been bred over decades and a vast amount of phenotypic data has been generated. In addition, recently whole genome sequencing-based genome-wide genotype data for many widely used inbred strains has been released. Here, we present an approach for in silico fine-mapping that uses genotypic data of 37 inbred mouse strains together with phenotypic data provided by the user to propose candidate variants and genes for the phenotype under study. Public genome-wide genotype data covering more than 74 million variant sites is queried efficiently in real-time to provide those variants that are compatible with the observed phenotype differences between strains. Variants can be filtered by molecular consequences and by corresponding molecular impact. Candidate gene lists can be generated from variant lists on the fly. Fine-mapping together with annotation or filtering of results is provided in a Bioconductor package called MouseFM. In order to characterize candidate variant lists under various settings, MouseFM was applied to two expression data sets across 20 inbred mouse strains, one from neutrophils and one from CD4+ T cells. Fine-mapping was assessed for about 10,000 genes, respectively, and identified candidate variants and haplotypes for many expression quantitative trait loci (eQTLs) reported previously based on these data. For albinism, MouseFM reports only one variant allele of moderate or high molecular impact that only albino mice share: a missense variant in the Tyr gene, reported previously to be causal for this phenotype. Performing in silico fine-mapping for interfrontal bone formation in mice using four strains with and five strains without interfrontal bone results in 12 genes. Of these, three are related to skull shaping abnormality. Finally performing fine-mapping for dystrophic cardiac calcification by comparing 9 strains showing the phenotype with eight strains lacking it, we identify only one moderate impact variant in the known causal gene Abcc6. In summary, this illustrates the benefit of using MouseFM for candidate variant and gene identification.

Dataset Information

Palatal segment contributions to midfacial growth in three inbred mouse strains.

Publications

Palatal segment contributions to midfacial anterior-posterior growth.

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