دورية أكاديمية
أعرض في EDS

Essential role for fibrillin-2 in zebrafish notochord and vascular morphogenesis.

التفاصيل البيبلوغرافية
العنوان: Essential role for fibrillin-2 in zebrafish notochord and vascular morphogenesis.
المؤلفون: Gansner JM; Department of Pediatrics, Washington University School of Medicine, St. Louis, Missouri 63110, USA., Madsen EC, Mecham RP, Gitlin JD
المصدر: Developmental dynamics : an official publication of the American Association of Anatomists [Dev Dyn] 2008 Oct; Vol. 237 (10), pp. 2844-61.
نوع المنشور: Journal Article; Research Support, N.I.H., Extramural
اللغة: English
بيانات الدورية: Publisher: Wiley Country of Publication: United States NLM ID: 9201927 Publication Model: Print Cited Medium: Print ISSN: 1058-8388 (Print) Linking ISSN: 10588388 NLM ISO Abbreviation: Dev Dyn Subsets: MEDLINE
أسماء مطبوعة: Original Publication: New York, NY : Wiley, c1992-
مواضيع طبية MeSH: Microfilament Proteins/*metabolism , Morphogenesis/*genetics , Notochord/*embryology , Notochord/*metabolism , Zebrafish/*embryology , Zebrafish/*metabolism, Animals ; Animals, Genetically Modified ; Embryo, Nonmammalian/blood supply ; Embryo, Nonmammalian/embryology ; Embryo, Nonmammalian/metabolism ; Fibrillins ; Gene Expression Regulation, Developmental ; Microfilament Proteins/deficiency ; Microfilament Proteins/genetics ; Microscopy, Electron, Transmission ; Mutation/genetics ; Notochord/blood supply ; Notochord/ultrastructure ; Phenotype ; Protein-Lysine 6-Oxidase/antagonists & inhibitors ; Protein-Lysine 6-Oxidase/metabolism ; Zebrafish/genetics
مستخلص: Recent studies demonstrate that lysyl oxidase cuproenzymes are critical for zebrafish notochord formation, but the molecular mechanisms of copper-dependent notochord morphogenesis are incompletely understood. We, therefore, conducted a forward genetic screen for zebrafish mutants that exhibit notochord sensitivity to lysyl oxidase inhibition, yielding a mutant with defects in notochord and vascular morphogenesis, puff daddygw1 (pfdgw1). Meiotic mapping and cloning reveal that the pfdgw1 phenotype results from disruption of the gene encoding the extracellular matrix protein fibrillin-2, and the spatiotemporal expression of fibrillin-2 is consistent with the pfdgw1 phenotype. Furthermore, each aspect of the pfdgw1 phenotype is recapitulated by morpholino knockdown of fibrillin-2. Taken together, the data reveal a genetic interaction between fibrillin-2 and the lysyl oxidases in notochord formation and demonstrate the importance of fibrillin-2 in specific early developmental processes in zebrafish.
(Copyright (c) 2008 Wiley-Liss, Inc.)
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معلومات مُعتمدة: T32 GM07200 United States GM NIGMS NIH HHS; T32 HD060554 United States HD NICHD NIH HHS; T32 GM007200 United States GM NIGMS NIH HHS; T32 HD060554-01 United States HD NICHD NIH HHS; DK44464 United States DK NIDDK NIH HHS; R01 DK044464 United States DK NIDDK NIH HHS; R37 DK044464 United States DK NIDDK NIH HHS
المشرفين على المادة: 0 (Fibrillins)
0 (Microfilament Proteins)
EC 1.4.3.13 (Protein-Lysine 6-Oxidase)
تواريخ الأحداث: Date Created: 20080926 Date Completed: 20081126 Latest Revision: 20181113
رمز التحديث: 20221213
مُعرف محوري في PubMed: PMC3081706
DOI: 10.1002/dvdy.21705
PMID: 18816837
قاعدة البيانات: MEDLINE
الوصف
تدمد:1058-8388
DOI:10.1002/dvdy.21705