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Neonatal Lethality of LGR5 Null Mice Is Associated with Ankyloglossia and Gastrointestinal Distension
Open accessAbstract
The physiological role of an orphan G protein-coupled receptor, LGR5, was investigated by targeted deletion of this seven-transmembrane protein containing a large N-terminal extracellular domain with leucine-rich repeats. LGR5 null mice exhibited 100% neonatal lethality characterized by gastrointestinal tract dilation with air and an absence of milk in the stomach. Gross and histological examination revealed fusion of the tongue to the floor of oral cavity in the mutant newborns and immunostaining of LGR5 expression in the epithelium of the tongue and in the mandible of the wild-type embryos. The observed ankyloglossia phenotype provides a model for understanding the genetic basis of this craniofacial defect in humans and an opportunity to elucidate the physiological role of the LGR5 signaling system during embryonic development.
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Cited by
Identification of stem cells in small intestine and colon by marker gene Lgr5
Barker, N., van Es, J. H., Kuipers, J., Kujala, P., van den Born, M., Cozijnsen, M., Haegebarth, A., Korving, J., Begthel, H., Peters, P. J., et al. (2007) · Nature · doi:10.1038/nature06196 · Cited by 5,687
Carmon, K. S., Gong, X., Lin, Q., Thomas, A., & Liu, Q. (2011) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.1106083108 · Cited by 855
The R-spondin/Lgr5/Rnf43 module: regulator of Wnt signal strength
de Lau, W., Peng, W. C., Gros, P., & Clevers, H. (2014) · Genes & Development · doi:10.1101/gad.235473.113 · Cited by 656
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Metadata
- Authors
- Morita, H., Mazerbourg, S., Bouley, D. M., Luo, C.-W., Kawamura, K., Kuwabara, Y., Baribault, H., Tian, H., & Hsueh, A. J. W.
- Venue
- Molecular and Cellular Biology
- Publisher
- Informa UK Limited
- Published
- 2004-11-01
- Volume
- 24
- Issue
- 22
- Pages
- 9736-9743
- ISSN
- 0270-7306 (ISSN-L)
- 1098-5549
- OpenAlex
- W1976206175
- Cited by
- 187
- References
- 0
- FWCI
- 14.12
- Type
- journal article
- Language
- en
- Open access
- green
- Licence
- journals.asm.org/non-commercial-tdm-license (tdm)
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- CoChat. (2026). Citation check: Neonatal Lethality of LGR5 Null Mice Is Associated with Ankyloglossia and Gastrointestinal Distension. https://app.cochat.ai/papers/10.1128/mcb.24.22.9736-9743.2004
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- Morita, H., Mazerbourg, S., Bouley, D. M., Luo, C.-W., Kawamura, K., Kuwabara, Y., Baribault, H., Tian, H., & Hsueh, A. J. W. (2004). Neonatal Lethality of LGR5 Null Mice Is Associated with Ankyloglossia and Gastrointestinal Distension. Molecular and Cellular Biology. https://doi.org/10.1128/mcb.24.22.9736-9743.2004
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