PMID: 9531541May 9, 1998Paper

Defects in limb, craniofacial, and thymic development in Jagged2 mutant mice

Genes & Development
R JiangT Gridley

Abstract

The Notch signaling pathway is a conserved intercellular signaling mechanism that is essential for proper embryonic development in numerous metazoan organisms. We have examined the in vivo role of the Jagged2 (Jag2) gene, which encodes a ligand for the Notch family of transmembrane receptors, by making a targeted mutation that removes a domain of the Jagged2 protein required for receptor interaction. Mice homozygous for this deletion die perinatally because of defects in craniofacial morphogenesis. The mutant homozygotes exhibit cleft palate and fusion of the tongue with the palatal shelves. The mutant mice also exhibit syndactyly (digit fusions) of the fore- and hindlimbs. The apical ectodermal ridge (AER) of the limb buds of the mutant homozygotes is hyperplastic, and we observe an expanded domain of Fgf8 expression in the AER. In the foot plates of the mutant homozygotes, both Bmp2 and Bmp7 expression and apoptotic interdigital cell death are reduced. Mutant homozygotes also display defects in thymic development, exhibiting altered thymic morphology and impaired differentiation of gamma delta lineage T cells. These results demonstrate that Notch signaling mediated by Jag2 plays an essential role during limb, craniofacial, an...Continue Reading

References

Dec 1, 1995·Nature Genetics·G ProetzelT Doetschman
Sep 28, 1995·Nature·S JarriaultA Israel
Mar 24, 1995·Cell·C E LindsellG Weinmaster
Apr 14, 1995·Science·S Artavanis-TsakonasM E Fortini
Dec 1, 1994·Developmental Biology·M A Muskavitch
Mar 15, 1994·Genes & Development·P J SwiatekT Gridley
Oct 21, 1994·Cell·M E Fortini, S Artavanis-Tsakonas
Jan 7, 1993·Nature·L Niswander, G R Martin
Feb 20, 1996·Proceedings of the National Academy of Sciences of the United States of America·R KopanJ S Nye
Jan 1, 1996·Developmental Genetics·C HofmannG Karsenty
Nov 25, 1996·Developmental Biology·C ShawberG Weinmaster
May 13, 1997·Proceedings of the National Academy of Sciences of the United States of America·S R Diehl, R P Erickson
Jul 1, 1997·Nature Genetics·T OdaS C Chandrasekharappa
Jan 1, 1997·Molecular and Cellular Neurosciences·G Weinmaster
Jan 1, 1997·Molecular and Cellular Neurosciences·T Gridley
Aug 1, 1997·Current Opinion in Genetics & Development·E Robey
Oct 10, 1997·Cell·R L Johnson, C J Tabin
Jan 1, 1997·Mammalian Genome : Official Journal of the International Mammalian Genome Society·Y LanT Gridley

❮ Previous
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Citations

Oct 21, 1999·Journal of Cellular Physiology·L Miele, B Osborne
Jul 4, 2002·International Journal of Hematology·K OhishiI D Bernstein
Oct 18, 2011·Calcified Tissue International·Stefano Zanotti, Ernesto Canalis
Dec 31, 2009·Apoptosis : an International Journal on Programmed Cell Death·Juan A Montero, Juan M Hurlé
Aug 8, 2013·Journal of Assisted Reproduction and Genetics·Gamze TanriverdiTulay Irez
Mar 17, 2004·Current Opinion in Immunology·Freddy RadtkeH Robson MacDonald
Mar 16, 2005·Current Opinion in Immunology·Daniel J PenningtonAdrian C Hayday
Sep 27, 2003·Developmental Biology·Daniel VasiliauskasClaudio D Stern
Sep 28, 2002·Trends in Genetics : TIG·Anna V Anagnostopoulos
Sep 5, 2001·Experimental Hematology·S Kojika, J D Griffin
Mar 14, 2000·Current Opinion in Immunology·M L Deftos, M J Bevan
Oct 6, 1999·Current Opinion in Neurobiology·J Y Wu, Y Rao
Apr 12, 2003·Seminars in Immunology·Warren S Pear, Freddy Radtke
Apr 12, 2003·Seminars in Immunology·Leonor ParreiraSérgio Simões
Apr 12, 2003·Seminars in Immunology·Ute KochCynthia J Guidos
Nov 30, 2002·Seminars in Immunology·Cynthia J Guidos
Mar 26, 2003·Seminars in Cell & Developmental Biology·Gabriela Hernández-Hoyos, José Alberola-Ila
Mar 22, 2002·Nature Reviews. Immunology·G Anderson, E J Jenkinson
Feb 27, 2001·Nature Immunology·E V Rothenberg
Feb 27, 2004·Nature Immunology·Freddy RadtkeH Robson MacDonald
Jan 24, 2007·Nature Immunology·Adrian C Hayday, Daniel J Pennington
Apr 19, 2007·Nature Immunology·Kenji Tanigaki, Tasuku Honjo
Apr 14, 2006·Nature Reviews. Cancer·Clemens GrabherA Thomas Look
Oct 3, 2001·The Journal of Experimental Medicine·A C JalecoL Parreira
Jan 31, 2007·The Journal of Experimental Medicine·Valerie BesseyriasFreddy Radtke
Oct 1, 2008·The Journal of Experimental Medicine·Katsuto HozumiSonoko Habu
Apr 10, 1999·Human Molecular Genetics·Y XueT Gridley
Dec 17, 2009·Human Molecular Genetics·Bryan C BjorkDavid R Beier
Mar 1, 2012·Human Molecular Genetics·Noriko FunatoHiromi Yanagisawa
Mar 6, 2003·Plastic and Reconstructive Surgery·Brian R GastmanErnest K Manders
Feb 23, 2012·Cold Spring Harbor Perspectives in Medicine·Jennifer J TungJan Kitajewski
Mar 19, 2005·Genes & Development·Allison E FalenderJoAnne S Richards
Dec 16, 1998·Immunological Reviews·S M Hedrick, L L Sharp
Dec 10, 2009·Molecular and Cellular Biology·Stefano Zanotti, Ernesto Canalis
May 18, 2007·Annual Review of Cell and Developmental Biology·Maria Ciofani, Juan Carlos Zúñiga-Pflücker
Nov 6, 2009·Annual Review of Genetics·Barbara Conradt

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