Dataset Information

When whorls collide: the development of hair patterns in frizzled 6 mutant mice.

ABSTRACT: Surface appendages such as bristles, feathers and hairs exhibit both long- and short-range order. In the frizzled 6 null (Fz6(-/-)) mouse the orientations of the earliest born hair follicles are uncorrelated, but over time the follicles reorient to create patterns that are characterized by a high degree of local order. By quantifying follicle orientations over time, in both living and fixed tissues, we define the time course of local hair follicle refinement and the resulting evolution of a montage of competing patterns in Fz6(-/-) skin. We observe an apparently local process that within one week can organize a field of many tens of thousands of follicles, generating long-range order that extends over distances of more than one centimeter. Physical systems that undergo an analogous ordering of vector components suggest potential mechanisms that might apply to the patterning of hair follicles and related biological structures.

SUBMITTER: Wang Y

PROVIDER: S-EPMC2976288 | biostudies-literature | 2010 Dec

REPOSITORIES: biostudies-literature

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When whorls collide: the development of hair patterns in frizzled 6 mutant mice.

Wang Yanshu Y Chang Hao H Nathans Jeremy J

Development (Cambridge, England) 20101201 23

Surface appendages such as bristles, feathers and hairs exhibit both long- and short-range order. In the frizzled 6 null (Fz6(-/-)) mouse the orientations of the earliest born hair follicles are uncorrelated, but over time the follicles reorient to create patterns that are characterized by a high degree of local order. By quantifying follicle orientations over time, in both living and fixed tissues, we define the time course of local hair follicle refinement and the resulting evolution of a mont ...[more]

PMID: 21062866

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Project description:BackgroundDifferences in cranial morphology arise due to changes in fundamental cell processes like migration, proliferation, differentiation and cell death driven by genetic programs. Signaling between fibroblast growth factors (FGFs) and their receptors (FGFRs) affect these processes during head development and mutations in FGFRs result in congenital diseases including FGFR-related craniosynostosis syndromes. Current research in model organisms focuses primarily on how these mutations change cell function local to sutures under the hypothesis that prematurely closing cranial sutures contribute to skull dysmorphogenesis. Though these studies have provided fundamentally important information contributing to the understanding of craniosynostosis conditions, knowledge of changes in cell function local to the sutures leave change in overall three-dimensional cranial morphology largely unexplained. Here we investigate growth of the skull in two inbred mouse models each carrying one of two gain-of-function mutations in FGFR2 on neighboring amino acids (S252W and P253R) that in humans cause Apert syndrome, one of the most severe FGFR-related craniosynostosis syndromes. We examine late embryonic skull development and suture patency in Fgfr2 Apert syndrome mice between embryonic day 17.5 and birth and quantify the effects of these mutations on 3D skull morphology, suture patency and growth.ResultsWe show in mice what studies in humans can only infer: specific cranial growth deviations occur prenatally and worsen with time in organisms carrying these FGFR2 mutations. We demonstrate that: 1) distinct skull morphologies of each mutation group are established by E17.5; 2) cranial suture patency patterns differ between mice carrying these mutations and their unaffected littermates; 3) the prenatal skull grows differently in each mutation group; and 4) unique Fgfr2-related cranial morphologies are exacerbated by late embryonic growth patterns.ConclusionsOur analysis of mutation-driven changes in cranial growth provides a previously missing piece of knowledge necessary for explaining variation in emergent cranial morphologies and may ultimately be helpful in managing human cases carrying these same mutations. This information is critical to the understanding of craniofacial development, disease and evolution and may contribute to the evaluation of incipient therapeutic strategies.

Dataset Information

When whorls collide: the development of hair patterns in frizzled 6 mutant mice.

Publications

When whorls collide: the development of hair patterns in frizzled 6 mutant mice.

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