Identification of Astrotactin2 as a Genetic Modifier That Regulates the Global Orientation of Mammalian Hair Follicles.
Autor: | Hao Chang, Hugh Cahill, Philip M Smallwood, Yanshu Wang, Jeremy Nathans |
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Jazyk: | angličtina |
Rok vydání: | 2015 |
Předmět: | |
Zdroj: | PLoS Genetics, Vol 11, Iss 9, p e1005532 (2015) |
Druh dokumentu: | article |
ISSN: | 1553-7390 1553-7404 |
DOI: | 10.1371/journal.pgen.1005532 |
Popis: | Planar cell polarity (PCP) signaling controls the global orientation of surface structures, such as hairs and bristles, in both vertebrates and invertebrates. In Frizzled6(-/-) (Fz6(-/-)) mice, hair follicle orientations on the head and back are nearly random at birth, but reorient during early postnatal development to eventually generate a nearly parallel anterior-to-posterior array. We report the identification of a naturally occurring exon 5 deletion in Astrotactin2 (Astn2) that acts as a recessive genetic modifier of the Fz6(-/-) hair patterning phenotype. A genetically engineered Astn2 exon 5 deletion recapitulates the modifier phenotype. In Fz6(-/-);Astn2(ex5del/del) mice, hair orientation on the back is subtly biased from posterior-to-anterior, leading to a 180-degree orientation reversal in mature mice. These experiments suggest that Astn2, an endosomal membrane protein, modulates PCP signaling. |
Databáze: | Directory of Open Access Journals |
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