title: Angora mouse mutation: altered hair cycle, follicular dystrophy, phenotypic maintenance of skin grafts, and changes in keratin expression.
abstract: Angora is an autosomal recessive mouse mutation caused by a deletion of approximately 2 kilobases in the fibroblast growth factor 5 (Fgf5) gene. Phenotypically, homozygous angora (Fgf5go/Fgf5go) mice have excessively long truncal hair and can be differentiated from heterozygous (+/Fgf5go) and wild-type (+/+) littermates by 21 days of age. Abnormal hair length is due to a prolongation of the anagen phase of the hair cycle of approximately 3 days. In addition, widely scattered hair follicles produce structurally defective hair shafts that twist within the follicle, presumably causing secondary hyperplasia of the outer root sheath and epidermis adjacent to the follicle. These follicular abnormalities were accentuated by immunohistochemical detection of mouse specific keratin 6, a nonspecific marker of epidermal hyperplasia. These abnormalities could be identified from birth throughout life in angora mice genotyped by polymerase chain reaction techniques. Moreover, the long truncal hair phenotype was maintained in skin grafted onto C.B-17/Sz-scid/scid mice that had normal pelage hairs and hair cycles, suggesting that circulating or diffusible humoral factors regulating the mouse hair cycle are not involved in this mutation. The angora mutation provides another useful mouse model for studying the hair cycle and its modulation.
authors.affiliation: Jackson Laboratory, Bar Harbor, ME, USA.
authors.forename: J P
authors.initials: JP
authors.lastname: Sundberg
authors.forename: M H
authors.initials: MH
authors.lastname: Rourk
authors.forename: D
authors.initials: D
authors.lastname: Boggess
authors.forename: M E
authors.initials: ME
authors.lastname: Hogan
authors.forename: B A
authors.initials: BA
authors.lastname: Sundberg
authors.forename: A P
authors.initials: AP
authors.lastname: Bertolino
mesh_terms: D000818:Animals; D000831:Animals, Newborn; D002453:Cell Cycle; D005260:Female; D051521:Fibroblast Growth Factor 5; D005346:Fibroblast Growth Factors / Q000235:genetics; D006085:Graft Survival / Q000235:genetics*; D006197:Hair / Q000254:growth & development* / Q000473:pathology*; D018859:Hair Follicle / Q000473:pathology*; D006720:Homozygote; D007633:Keratins / Q000096:biosynthesis*; D008297:Male; D051379:Mice; D008810:Mice, Inbred C57BL; D008817:Mice, Mutant Strains / Q000235:genetics*; D016513:Mice, SCID; D009154:Mutation; D010641:Phenotype; D017384:Sequence Deletion; D016038:Skin Transplantation / Q000473:pathology*
chemical_list: C496450:Fgf5 protein, mouse; D051521:Fibroblast Growth Factor 5; D005346:Fibroblast Growth Factors; D007633:Keratins