Angora mouse mutation: Altered hair cycle, follicular dystrophy, phenotypic maintenance of skin grafts, and changes in keratin expression

Angora mouse mutation: Altered hair cycle, follicular dystrophy, phenotypic maintenance of skin grafts, and changes in keratin expression
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DOI:
10.1177/030098589703400301
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发表时间:
1997-05-01
影响因子:
2.4
通讯作者:
Bertolino, AP
Bertolino, AP
中科院分区:
农林科学2区
文献类型:
--
作者:
Sundberg, JP;Rourk, MH;Bertolino, AP

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安哥拉是一种常染色体隐性小鼠突变,由成纤维细胞生长因子5(Fgf 5)基因中的2个类似酶缺失引起。表型上,纯合安哥拉(Fgf 5(go)/FgfS(go))小鼠具有过长的干毛,并且可以在21日龄时与杂合(+/Fgf 5(go))和野生型(+/+)同窝仔区分开。毛发长度异常是由于毛发生长周期的生长期延长约3天。此外,广泛分散的毛囊产生结构上有缺陷的毛干,毛干在毛囊内扭曲,可能导致毛囊附近的外根鞘和表皮的继发性增生。这些滤泡异常加重免疫组化检测小鼠特异性角蛋白6,表皮增生的非特异性标志物。通过聚合酶链反应技术对安哥拉小鼠进行基因分型,这些异常可以从出生到整个生命过程中识别出来。此外,在移植到具有正常被毛和毛发周期的C.B-17/S-z-scid/scid小鼠的皮肤中保持了长干毛表型,这表明调节小鼠毛发周期的循环或可扩散体液因子不参与该突变。安哥拉山羊的突变为研究毛发周期及其调节提供了另一种有用的小鼠模型。
Angora is an autosomal recessive mouse mutation caused by a deletion of similar to 2 kilobases in the fibroblast growth factor 5 (Fgf5) gene. Phenotypically, homozygous angora (Fgf5(go)/FgfS(go)) mice have excessively long truncal hair and can be differentiated from heterozygous (+/Fgf5(go)) 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/S-z-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.