دورية أكاديمية
Laminin γ2 knockout mice rescued with the human protein exhibit enamel maturation defects.
| العنوان: | Laminin γ2 knockout mice rescued with the human protein exhibit enamel maturation defects. |
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| المؤلفون: | Wazen RM; Laboratory for the Study of Calcified Tissues and Biomaterials, Department of Stomatology, Faculty of Dentistry, Université de Montréal, Montréal, Québec, Canada., Viegas-Costa LC; Laboratory for the Study of Calcified Tissues and Biomaterials, Department of Stomatology, Faculty of Dentistry, Université de Montréal, Montréal, Québec, Canada., Fouillen A; Laboratory for the Study of Calcified Tissues and Biomaterials, Department of Stomatology, Faculty of Dentistry, Université de Montréal, Montréal, Québec, Canada; Department of Biochemistry and Molecular Medicine, Université de Montréal, Montréal, Québec, Canada., Moffatt P; Shriners Hospital for Children, Montréal, Montréal, Québec, Canada., Adair-Kirk TL; Department of Medicine, Washington University School of Medicine, St. Louis, MO., Senior RM; Department of Medicine, Washington University School of Medicine, St. Louis, MO., Nanci A; Laboratory for the Study of Calcified Tissues and Biomaterials, Department of Stomatology, Faculty of Dentistry, Université de Montréal, Montréal, Québec, Canada; Department of Biochemistry and Molecular Medicine, Université de Montréal, Montréal, Québec, Canada. Electronic address: antonio.nanci@umontreal.ca. |
| المصدر: | Matrix biology : journal of the International Society for Matrix Biology [Matrix Biol] 2016 May-Jul; Vol. 52-54, pp. 207-218. Date of Electronic Publication: 2016 Mar 05. |
| نوع المنشور: | Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't |
| اللغة: | English |
| بيانات الدورية: | Publisher: Elsevier Country of Publication: Netherlands NLM ID: 9432592 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1569-1802 (Electronic) Linking ISSN: 0945053X NLM ISO Abbreviation: Matrix Biol Subsets: MEDLINE |
| أسماء مطبوعة: | Publication: Amsterdam : Elsevier Original Publication: Stuttgart ; New York : Fischer, c1994- |
| مواضيع طبية MeSH: | Basement Membrane/*pathology , Enamel Organ/*ultrastructure , Laminin/*genetics , Laminin/*metabolism, Amelogenesis ; Animals ; Basement Membrane/cytology ; Cell Differentiation ; Enamel Organ/cytology ; Genes, Lethal ; Humans ; Incisor ; Mice ; Mice, Knockout ; Mice, Transgenic ; X-Ray Microtomography |
| مستخلص: | The epithelial ameloblasts are separated from the maturing enamel by an atypical basement membrane (BM) that is enriched in laminin 332 (LM-332). This heterotrimeric protein (α3, ß3 and γ2 chains) provides structural integrity to BMs and influences various epithelial cell processes including cell adhesion and differentiation. Mouse models that lack expression of individual LM-332 chains die shortly after birth. The lethal phenotype of laminin γ2 knockout mice can be rescued by human laminin γ2 (LAMC2) expressed using a doxycycline-inducible (Tet-on) cytokeratin 14 promoter-rtTA. These otherwise normal-looking rescued mice exhibit white spot lesions on incisors. We therefore investigated the effect of rescue with human LAMC2 on enamel maturation and structuring of the atypical BM. The maturation stage enamel organ in transgenic mice was severely altered as compared to wild type controls, a structured BM was no longer discernible, dystrophic matrix appeared in the maturing enamel layer, and there was residual enamel matrix late into the maturation stage. Microtomographic scans revealed excessive wear of occlusal surfaces on molars, chipping of enamel on incisor tips, and hypomineralization of the enamel layer. No structural alterations were observed at other epithelial sites, such as skin, palate and tongue. These results indicate that while this humanized mouse model is capable of rescue in various epithelial tissues, it is unable to sustain structuring of a proper BM at the interface between ameloblasts and maturing enamel. This failure may be related to the atypical composition of the BM in the maturation stage and reaffirms that the atypical BM is essential for enamel maturation. (Copyright © 2016 International Society of Matrix Biology. Published by Elsevier B.V. All rights reserved.) |
| معلومات مُعتمدة: | MOP110972 Canada CIHR |
| فهرسة مساهمة: | Keywords: Amelogenesis; Basement membrane; Enamel maturation; Humanized mouse model; Laminin γ2 |
| المشرفين على المادة: | 0 (LAMC2 protein, human) 0 (Lamc2 protein, mouse) 0 (Laminin) |
| تواريخ الأحداث: | Date Created: 20160310 Date Completed: 20171220 Latest Revision: 20180103 |
| رمز التحديث: | 20240628 |
| DOI: | 10.1016/j.matbio.2016.03.002 |
| PMID: | 26956061 |
| قاعدة البيانات: | MEDLINE |
Full Text Finder | تدمد: | 1569-1802 |
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| DOI: | 10.1016/j.matbio.2016.03.002 |