Shirokova, Vera; Jussila, Maria; Hytönen, Marjo K.; Perälä, Nina; Drögemüller, Cord; Leeb, Tosso; Lohi, Hannes; Sainio, Kirsi; Thesleff, Irma; Mikkola, Marja L. (2013). Expression of Foxi3 is regulated by ectodysplasin in skin appendage placodes. Developmental dynamics, 242(6), pp. 593-603. John Wiley & Sons 10.1002/dvdy.23952
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BACKGROUND
Foxi3 is a member of the large forkhead box family of transcriptional regulators, which have a wide range of biological activities including manifold developmental processes. Heterozygous mutation in Foxi3 was identified in several hairless dog breeds characterized by sparse fur coat and missing teeth. A related phenotype called hypohidrotic ectodermal dysplasia (HED) is caused by mutations in the ectodysplasin (Eda) pathway genes.
RESULTS
Expression of Foxi3 was strictly confined to the epithelium in developing ectodermal appendages in mouse embryos, but no expression was detected in the epidermis. Foxi3 was expressed in teeth and hair follicles throughout embryogenesis, but in mammary glands only during the earliest stages of development. Foxi3 expression was decreased and increased in Eda loss- and gain-of-function embryos, respectively, and was highly induced by Eda protein in embryonic skin explants. Also activin A treatment up-regulated Foxi3 mRNA levels in vitro.
CONCLUSIONS
Eda and activin A were identified as upstream regulators of Foxi3. Foxi3 is a likely transcriptional target of Eda in ectodermal appendage placodes suggesting that HED phenotype may in part be produced by compromised Foxi3 activity. In addition to hair and teeth, Foxi3 may have a role in nail, eye, and mammary, sweat, and salivary gland development.
Item Type: |
Journal Article (Original Article) |
|---|---|
Division/Institute: |
05 Veterinary Medicine > Research Foci > DermFocus 05 Veterinary Medicine > Department of Clinical Research and Veterinary Public Health (DCR-VPH) > Institute of Genetics 05 Veterinary Medicine > Department of Clinical Research and Veterinary Public Health (DCR-VPH) |
UniBE Contributor: |
Drögemüller, Cord, Leeb, Tosso |
Subjects: |
500 Science > 590 Animals (Zoology) 600 Technology > 630 Agriculture |
ISSN: |
1058-8388 |
Publisher: |
John Wiley & Sons |
Language: |
English |
Submitter: |
Susanne Portner |
Date Deposited: |
22 Aug 2014 12:13 |
Last Modified: |
05 Dec 2022 14:29 |
Publisher DOI: |
10.1002/dvdy.23952 |
PubMed ID: |
23441037 |
Uncontrolled Keywords: |
Foxi3, ectodermal dysplasia, Edar, tooth development, hair follicle development |
BORIS DOI: |
10.7892/boris.43884 |
URI: |
https://boris.unibe.ch/id/eprint/43884 |
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