Expression of Foxi3 is regulated by ectodysplasin in skin appendage placodes

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 and 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:

14 Oct 2015 11:34

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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