The DOI matches its Crossref record; 27 of the 27 references it cites were found in a registry or an index.
Control of Skeletal Patterning by EphrinB1-EphB Interactions
Open accessAbstract
We report that targeted inactivation of the Eph receptor ligand ephrinB1 in mouse caused perinatal lethality, edema, defective body wall closure, and skeletal abnormalities. In the thorax, sternocostal connections were arranged asymmetrically and sternebrae were fused, defects that were phenocopied in EphB2/EphB3 receptor mutants. In the wrist, loss of ephrinB1 led to abnormal cartilage segmentation and the formation of additional skeletal elements. We conclude that ephrinB1 and B class Eph receptors provide positional cues required for the normal morphogenesis of skeletal elements. Another malformation, preaxial polydactyly, was exclusively seen in heterozygous females in which expression of the X-linked ephrinB1 gene was mosaic, so that ectopic EphB-ephrinB1 interactions led to restricted cell movements and the bifurcation of digital rays. Our findings suggest that differential cell adhesion and sorting might be relevant for an unusual class of X-linked human genetic disorders, in which heterozygous females show more severe phenotypes than hemizygous males.
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“In the female, random X-inactivation generates groups of cells that either express, or do not express ephrin-B1, and the homophilic sorting process increases the patch size of these cells, causing abnormal tissue boundaries (for example, in the coronal sutures and limbs).33”
From Craniosynostosis (2011) · cites this paper as [33]
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Cites
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Metadata
- Authors
- Compagni, A., Logan, M., Klein, R., & Adams, R. H.
- Affiliations
- Cancer Research UK (GB)
- Max Planck Society (DE)
- Corresponding author
- Adams, Ralf H.
- Venue
- Developmental Cell
- Publisher
- Elsevier BV
- Published
- 2003-08
- Volume
- 5
- Issue
- 2
- Pages
- 217-230
- ISSN
- 1534-5807 (ISSN-L)
- OpenAlex
- W2014781971
- Cited by
- 231
- References
- 27
- Citations by year
- 2025: 3 · 2024: 9 · 2023: 3 · 2022: 9 · 2021: 7 · 2020: 10 · 2019: 6 · 2018: 13 · 2017: 7 · 2016: 12 · 2015: 9 · 2014: 6 · 2013: 16 · 2012: 12
- FWCI
- 4.56
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Licence
- Locations
- Developmental Cell (publishedVersion)
- PubMed (publishedVersion)
- Max Planck Digital Library (submittedVersion)
- MPG.PuRe (Max Planck Society) (submittedVersion)
- Topics
- Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Axon Guidance and Neuronal Signaling
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Developmental Biology and Gene Regulation
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Developmental Biology › Congenital limb and hand anomalies
- Keywords
- ephrin-B1, EphB receptors, EphB2, EphB3, skeletal patterning, cartilage segmentation, preaxial polydactyly, X-linked disorders, positional cues, cell adhesion, cell sorting, heterozygous females, perinatal lethality, mouse mutants
- MeSH terms
- Congenital Abnormalities, Animals, Embryo, Mammalian, Female, Humans, Male, Morphogenesis, Phenotype, Skeleton, Sternum, Signal Transduction, In Situ Hybridization, Gene Targeting, Body Patterning, Receptors, Eph Family, Receptor, EphB2, Ephrin-B1, Mice
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