Orai1 expression pattern in tooth and craniofacial ectodermal tissues and potential functions during ameloblast differentiation.

Content Provider	Semantic Scholar
Author	Zheng, L. I. Zinn, Vina Lefkelidou, Anna Taqi, Nawar Chatzistavrou, Xanthippi Balam, Tarek Nervina, Jeanne Papagerakis, Silvana Papagerakis, Petros
Copyright Year	2015
Abstract	BACKGROUND Orai1 is a plasma membrane protein that forms the pore of the calcium release activated calcium channel. Humans with mutated Orai1 present with hereditary combined immunodeficiency, congenital myopathy and anhidrotic ectodermal dysplasia. Consistent with the ectodermal dysplasia phenotype, enamel formation and mineralization is also abnormal in Orai1 deficient patients. The expression pattern and potential functions of Orai1 in enamel formation remains unclear. To contribute toward understanding the role of Orai1 in amelogenesis we characterized ORAI1 protein developmental pattern in comparison with other ectodermal organs. We also examined the effects of Orai1 down-regulation in ameloblast cell proliferation and differentiation. RESULTS Our data show strong expression of ORAI1 protein during the ameloblast secretory stage, which weans at the end of the maturation stage. In salivary glands, ORAI1 is expressed mainly in acini cells. ORAI1 expression is also found in hair follicle and oral epithelium. Knockdown of Orai1 expression decreases cell proliferation and results in RNA expression levels changes of key ameloblast genes regulating enamel thickness and mineralization. CONCLUSIONS This study provides insights in the anhidrotic ectodermal dysplasia phenotype due to Orai1 mutation and highlights the importance of calcium signaling in controlling ameloblast differentiation and maturation during tooth development.
Starting Page	518
Ending Page	523
Page Count	6
File Format	PDF HTM / HTML
Alternate Webpage(s)	https://deepblue.lib.umich.edu/bitstream/handle/2027.42/113781/dvdy24307.pdf?isAllowed=y&sequence=1
PubMed reference number	26178077v1
Alternate Webpage(s)	https://doi.org/10.1002/dvdy.24307
DOI	10.1002/dvdy.24307
Journal	Developmental dynamics : an official publication of the American Association of Anatomists
Volume Number	244
Issue Number	10
Language	English
Access Restriction	Open
Subject Keyword	Ameloblasts Amelogenesis Anhydrotic Ectodermal Dysplasias Biologic Development Body tissue Calcification Calcium Channel Calcium Signaling Cell Differentiation process Cell Proliferation Cell secretion Congenital myopathy (disorder) Dental Enamel Down-Regulation Ectoderm Ectodermal Dysplasia Gland Hair follicle structure Immunologic Deficiency Syndromes Membrane Proteins Mutation ORAI1 gene Odontogenesis Odontogenic Tissue Organ Patients Plasma membrane Salivary Glands Transcription, Genetic X-Linked Combined Immunodeficiency Diseases ameloblast differentiation craniofacial negative regulation of hematopoietic progenitor cell differentiation
Content Type	Text
Resource Type	Article