Journal Article

Ectodysplasin-A1 is sufficient to rescue both hair growth and sweat glands in Tabby mice

Anand K. Srivastava, Meredith C. Durmowicz, Andrew J. Hartung, Joan Hudson, Lizbeth V. Ouzts, David M. Donovan, Chang-Yi Cui and David Schlessinger

in Human Molecular Genetics

Volume 10, issue 26, pages 2973-2981
Published in print December 2001 | ISSN: 0964-6906
Published online December 2001 | e-ISSN: 1460-2083 | DOI:
Ectodysplasin-A1 is sufficient to rescue both hair growth and sweat glands in Tabby mice

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Mutations in the human ectodysplasin-A (EDA) are responsible for the most common form of the ectodermal dysplasia and the defective orthologous gene in mice produces the tabby phenotype, suggesting its vital role in the development of hair, sweat glands and teeth. Among several EDA splice isoforms, the most common and the longest EDA splice isoforms, EDA-A1 and EDA-A2, differing by only two amino acids, activate NF-κB-promoted transcription by binding to distinct receptors, EDAR and XEDAR. The extent to which any particular isoform is sufficient for the formation of hair, sweat glands or teeth has remained unclear. Here we report that transgenic expression of the mouse EDA-A1 isoform in tabby (EDA-less) males rescued development of several skin appendages. The transgenic tabby mice showed almost complete restoration of hair growth, dermal ridges, sweat glands and molars. The number of hair follicles in the transgenic mice is the same as in wild-type; though the development of follicles and associated glands varies from indistinguishable from wild-type to smaller and/or only partially formed. These results suggest that the other EDA isoforms may not be absolutely required for skin appendage formation, but consistent with distinctive temporal and spatial expression of the EDA-A2 isoform, are likely required for appropriate timing and completeness of development. Our data provide the first direct physiological evidence that EDA-A1 is a key regulator of hair follicle and sweat gland initiation; its soluble ligand form could aid in deriving therapeutic reagents for conditions affecting hair and sweat gland formation.

Journal Article.  5763 words.  Illustrated.

Subjects: Genetics and Genomics

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