{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,22]],"date-time":"2026-10-22T14:11:38Z","timestamp":1792678298029,"version":"4.5.0"},"reference-count":53,"publisher":"The Company of Biologists","issue":"4","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2002,2,15]]},"abstract":"<jats:p>Alagille syndrome is a human autosomal dominant developmental disorder characterized by liver, heart, eye, skeletal, craniofacial and kidney abnormalities. Alagille syndrome is caused by mutations in the Jagged 1 (JAG1) gene, which encodes a ligand for Notch family receptors. The majority of JAG1 mutations seen in Alagille syndrome patients are null alleles, suggesting JAG1 haploinsufficiency as a primary cause of this disorder. Mice homozygous for a Jag1 null mutation die during embryogenesis and Jag1\/+ heterozygous mice exhibit eye defects but do not exhibit other phenotypes characteristic of Alagille syndrome patients (Xue, Y., Gao, X., Lindsell, C. E., Norton, C. R., Chang, B., Hicks, C., Gendron-Maguire, M., Rand, E. B., Weinmaster, G. and Gridley, T. (1999) Hum. Mol. Genet.8, 723-730). Here we report that mice doubly heterozygous for the Jag1 null allele and a Notch2 hypomorphic allele exhibit developmental abnormalities characteristic of Alagille syndrome. Double heterozygous mice exhibit jaundice, growth retardation, impaired differentiation of intrahepatic bile ducts and defects in heart, eye and kidney development. The defects in bile duct epithelial cell differentiation and morphogenesis in the double heterozygous mice are similar to defects in epithelial morphogenesis of Notch pathway mutants in Drosophila, suggesting that a role for the Notch signaling pathway in regulating epithelial morphogenesis has been conserved between insects and mammals. This work also demonstrates that the Notch2 and Jag1 mutations interact to create a more representative mouse model of Alagille syndrome and provides a possible explanation of the variable phenotypic expression observed in Alagille syndrome patients.<\/jats:p>","DOI":"10.1242\/dev.129.4.1075","type":"journal-article","created":{"date-parts":[[2021,4,23]],"date-time":"2021-04-23T16:27:43Z","timestamp":1619195263000},"page":"1075-1082","source":"Crossref","is-referenced-by-count":425,"title":["A mouse model of Alagille syndrome:\n                    <i>Notch2<\/i>\n                    as a genetic modifier of\n                    <i>Jag1<\/i>\n                    haploinsufficiency"],"prefix":"10.1242","volume":"129","author":[{"given":"Brent","family":"McCright","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Julie","family":"Lozier","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas","family":"Gridley","sequence":"additional","affiliation":[{"name":"The Jackson Laboratory, 600 Main Street, Bar Harbor, Maine 04609, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","reference":[{"key":"2021042513145785200_ALAGILLE-ETAL-1987","doi-asserted-by":"crossref","unstructured":"Alagille, D., Estrada, A., Hadchouel, M., Gautier, M., Odievre, M. and Dommergues, J. P. (1987). Syndromic paucity of interlobular bile ducts (Alagille syndrome or arteriohepatic dysplasia: Review of 80 cases. J. Pediatr.110, 195-200.","DOI":"10.1016\/S0022-3476(87)80153-1"},{"key":"2021042513145785200_ARTAVANIS-TSAKONAS-ETAL-1991","doi-asserted-by":"crossref","unstructured":"Artavanis-Tsakonas, S., Delidakis, C. and Fehon, R. G. (1991). The Notch locus and the cell biology of neuroblast segregation. Annu. Rev. Cell Biol.7, 427-452.","DOI":"10.1146\/annurev.cb.07.110191.002235"},{"key":"2021042513145785200_ARTAVANIS-TSAKONAS-ETAL-1999","doi-asserted-by":"crossref","unstructured":"Artavanis-Tsakonas, S., Rand, M. D. and Lake, R. J. (1999). Notch signaling: cell fate control and signal integration in development. Science284, 770-776.","DOI":"10.1126\/science.284.5415.770"},{"key":"2021042513145785200_BRAND-AND-CAMPOS-ORTEGA-1990","doi-asserted-by":"crossref","unstructured":"Brand, M. and Campos-Ortega, J. A. (1990). Second-site modifiers of the split mutation of Notch define genes involved in neurogenesis in Drosophila melanogaster. Roux\u2019s Arch. Dev. Biol. 198, 275-285.","DOI":"10.1007\/BF00377394"},{"key":"2021042513145785200_BULMAN-ETAL-2000","doi-asserted-by":"crossref","unstructured":"Bulman, M. P., Kusumi, K., Frayling, T. M., McKeown, C., Garrett, C., Lander, E. S., Krumlauf, R., Hattersley, A. T., Ellard, S. and Turnpenny, P. D. (2000). Mutations in the human delta homologue, DLL3, cause axial skeletal defects in spondylocostal dysostosis. Nature Genet.24, 438-441.","DOI":"10.1038\/74307"},{"key":"2021042513145785200_CORNELL-ETAL-1999","doi-asserted-by":"crossref","unstructured":"Cornell, M., Evans, D. A., Mann, R., Fostier, M., Flasza, M., Monthatong, M., Artavanis-Tsakonas, S. and Baron, M. (1999). The Drosophila melanogaster Suppressor of deltex gene, a regulator of the Notch receptor signaling pathway, is an E3 class ubiquitin ligase. Genetics152, 567-576.","DOI":"10.1093\/genetics\/152.2.567"},{"key":"2021042513145785200_COTRAN-ETAL-1999","unstructured":"Cotran, R. S., Kumar, V. and Collins, T. (1999). Robbins Pathologic Basis of Disease, 6th edition. Philadelphia: W.B. Saunders."},{"key":"2021042513145785200_CROSNIER-ETAL-2000","doi-asserted-by":"crossref","unstructured":"Crosnier, C., Attie-Bitach, T., Encha-Razavi, F., Audollent, S., Soudy, F., Hadchouel, M., Meunier-Rotival, M. and Vekemans, M. (2000). JAGGED1 gene expression during human embryogenesis elucidates the wide phenotypic spectrum of Alagille syndrome. Hepatology32, 574-581.","DOI":"10.1053\/jhep.2000.16600"},{"key":"2021042513145785200_DOBENS-AND-RAFTERY-2000","doi-asserted-by":"crossref","unstructured":"Dobens, L. L. and Raftery, L. A. (2000). Integration of epithelial patterning and morphogenesis in Drosophila ovarian follicle cells. Dev. Dyn. 218, 80-93.","DOI":"10.1002\/(SICI)1097-0177(200005)218:1<80::AID-DVDY7>3.0.CO;2-8"},{"key":"2021042513145785200_ELDADAH-ETAL-2001","doi-asserted-by":"crossref","unstructured":"Eldadah, Z. A., Hamosh, A., Biery, N. J., Montgomery, R. A., Duke, M., Elkins, R. and Dietz, H. C. (2001). Familial Tetralogy of Fallot caused by mutation in the jagged1 gene. Hum. Mol. Genet.10, 163-169.","DOI":"10.1093\/hmg\/10.2.163"},{"key":"2021042513145785200_EMERICK-ETAL-1999","doi-asserted-by":"crossref","unstructured":"Emerick, K. M., Rand, E. B., Goldmuntz, E., Krantz, I. D., Spinner, N. B. and Piccoli, D. A. (1999). Features of Alagille syndrome in 92 patients: frequency and relation to prognosis. Hepatology29, 822-829.","DOI":"10.1002\/hep.510290331"},{"key":"2021042513145785200_FULLER-ETAL-1989","doi-asserted-by":"crossref","unstructured":"Fuller, M. T., Regan, C. L., Green, L. L., Robertson, B., Deuring, R. and Hays, T. S. (1989). 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T., Daly, M. J., Reeve, M. P., Hawkins, T. L., Birren, B. W., Jaenisch, R. and Lander, E. S. (1997). Serrate2 is disrupted in the mouse limb development mutant syndactylism. Nature389, 722-725.","DOI":"10.1038\/39587"},{"key":"2021042513145785200_SPINNER-ETAL-2001","doi-asserted-by":"crossref","unstructured":"Spinner, N. B., Colliton, R. P., Crosnier, C., Krantz, I. D., Hadchouel, M. and Meunier-Rotival, M. (2001) Jagged1 mutations in alagille syndrome. Hum. Mutat. 17, 18-33.","DOI":"10.1002\/1098-1004(2001)17:1<18::AID-HUMU3>3.0.CO;2-T"},{"key":"2021042513145785200_STEARNS-AND-BOTSTEIN-1988","doi-asserted-by":"crossref","unstructured":"Stearns, T. and Botstein, D. (1988). Unlinked noncomplementation: isolation of new conditional-lethal mutations in each of the tubulin genes of Saccharomyces cerevisiae. 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The mouse slalom mutant demonstrates a role for Jagged1 in neuroepithelial patterning in the organ of Corti. Hum. Mol. Genet. 10, 507-512.","DOI":"10.1093\/hmg\/10.5.507"},{"key":"2021042513145785200_VERHEYEN-ETAL-1996","doi-asserted-by":"crossref","unstructured":"Verheyen, E. M., Purcell, K. J., Fortini, M. E. and Artavanis-Tsakonas, S. (1996). Analysis of dominant enhancers and suppressors of activated Notch in Drosophila. Genetics144, 1127-1141.","DOI":"10.1093\/genetics\/144.3.1127"},{"key":"2021042513145785200_VIJAYAN-AND-TAN-1992","unstructured":"Vijayan, V. and Tan, C. E. (1992). Developing human biliary system in three dimensions. Anat. Rec.249, 389-398."},{"key":"2021042513145785200_WEINMASTER-2000","doi-asserted-by":"crossref","unstructured":"Weinmaster, G. (2000). Notch signal transduction: a real Rip and more. Curr. Opin. Genet. 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