Many congenital malformations, such as cleft palate and neural tube defects, have a multifactorial origin involving both environmental and genetic factors. Conditions such as these may be exclusively monogenic, polygenic or environmental, but in most cases both genetic and environmental factors are involved1. This study describes the sub-chromosomal localization of a single gene defect causing cleft palate and ankyloglossia (tongue-tied) in a large Icelandic family. This defect is a model for the analysis of other neural-crest malformations that show a more complex multi-factorial inheritance pattern.
This is a preview of subscription content, access via your institution
Subscribe to this journal
Receive 51 print issues and online access
$199.00 per year
only $3.90 per issue
Rent or buy this article
Get just this article for as long as you need it
Prices may be subject to local taxes which are calculated during checkout
Thompson, J. S. & Thompson, M. W. Genetics in Medicine 4th edn (W. B. Saunders, Philadelphia, 1986).
Moore, K. L. The Developing Human 3rd edn (W. B. Saunders, Philadelphia, 1982).
Leek, I. Br. Med. Bull. 40, 390–395 (1984).
Witkop, C. J., MacCollum, D. W. & Rubin, A. in Handbook of Congenital Malformations (ed. Rubin, A.) 103–156 (W. B. Saunders, Philadelphia, 1967).
Burdi, A. R. & Silvey, R. G. Cleft Palate J. 6, 1–7 (1969).
Davies, K. E. et al. Nucleic Acids Res. 11, 2303–2312 (1983).
Royer-Pokura, B. et al. Nature 322, 32–38 (1986).
Gusella, J. et al. Nature 306, 234–238 (1983).
Knowlton, R. G. et al. Nature 318, 380–382 (1985).
Wainwright, B. J. et al. Nature 318, 384–385 (1985).
White, R. et al. Nature 318, 382–384 (1985).
Bjornsson, A. & Arnason, A. 19th Cong. Scand. Ass. Plastic Surgeons, Abstract, Gothenburg (1986).
Page, D. C., Harper, M. E., Love, J. & Botstein, D. Nature 311, 119–123 (1984).
Page, D. C. et al. Proc. natn. Acad. Sci. U.S.A. 79, 5352–5356 (1982).
Poustka, A. & Lehrach, H. Trends Genet. 2, 174–179 (1986).
Schwartz, D. C. & Cantor, C. R. Cell 37, 67–75 (1984).
Burn, J. & Gibbens, D. J. med. Genet. 16, 210–214 (1979).
Brown, M. S. & Goldstein, J. L. A. Rev. Biochem. 52, 233–261 (1983).
Bodmer, W. F. in Evolution from Molecules to Man (ed. Bendall, D. S.) 197–208 (Cambridge university Press, 1983).
Lathrop, G. M., Lalouel, J. M., Julier, C. & Ott, J. Proc. natn. Acad. Sci. U.S.A. 81, 3443–3446 (1984).
Ott, J. Analysis of Human Gentic Linkage 136–137 (Johns Hopkins University Press, Baltimore, 1985).
Goodfellow, P. N., Davies, K. E. & Ropers, H.-H. Cytogenet. Cell Genet. 40, 296–352 (1985).
Kunkel, L. M. et al. Proc. natn. Acad. Sci. U.S.A. 74, 1245–1249 (1977).
Southern, E. M. J. molec. Biol. 98, 503–517 (1975).
Rights and permissions
About this article
Cite this article
Moore, G., Ivens, A., Chambers, J. et al. Linkage of an X-chromosome cleft palate gene. Nature 326, 91–92 (1987). https://doi.org/10.1038/326091a0
This article is cited by
Tongue Tie in Infancy
Current Treatment Options in Pediatrics (2016)
The T-box transcription factor gene TBX22 is mutated in X-linked cleft palate and ankyloglossia
Nature Genetics (2001)
Time for T
Nature Genetics (2001)
Cloning and characterization of a novel bicoid-related homeobox transcription factor gene, RIEG, involved in Rieger syndrome
Nature Genetics (1996)
Genetics meets the press
Nature Genetics (1995)
By submitting a comment you agree to abide by our Terms and Community Guidelines. If you find something abusive or that does not comply with our terms or guidelines please flag it as inappropriate.