Abstract
The autosomal recessive gene hairless (hr) is responsible for the complete hairlessness in mice homozygous for this gene. Hair shedding that begins at the age of 10 days is caused by an abnormal cycle of hair follicle development disturbed at the catagen stage. This results in enhanced programmed cell death (apoptosis) and ultimately leads to the complete hair follicle destruction and shedding of all hairs by the age of three weeks. To study the phenotypic expression of the hr gene in a chimeric organism, we have obtained 12 chimeric mice hr/hr ↔ +/+ by means of aggregation of early embryos hr/hr and +/+. In chimeric mice, the hair shedding has begun two days later than in the hr/hr mice. By day 23 of postnatal development, hairless areas were present on the coat of chimeric mice or the latter were completely hairless depending on the percentage of the hr/hr mutant component. In four chimeras with high content of the mutant component (68–76%), the hair shedding process was similar to that in the hr/hr mice, though it was accomplished two days later. In three chimeras with 48–51% of the mutant component, alternating hairless and hair-covered bands were observed. These data suggest that the hr gene acts in epidermal cells of a hair follicle, because epidermal cell clones in embryonic skin migrate in the lateral–ventral direction coherently and without mixing. However, some chimeras displayed a pattern which was not so clear-cut: the band borders were illegible and hairs partly covered the hairless areas. In some chimeras, the uniform thinning of the coat was observed. Analysis of the effects of the hr mutant gene in chimeric mice differing in the ratio between mutant (hr/hr) and normal (+/+) components in tissues suggests that the hrgene acts in the epidermal cells of the hair follicle. The interactions between cells have an essential effect on the mode and degree of the hr gene expression, which leads to distortion of the “ectodermal” coat pattern in chimeras.
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References
Millar, S.E., Molecular Mechanisms Regulating Hair Follicle Development, J. Invest. Dermatol., 2002, vol. 118, pp. 216-225.
Panteleyev, A.A., Jahoda, C.A.B., and Christiano, A.M., Hair Follicle Predetermination, J. Cell Sci., 2001, vol. 114, pp. 3419-3431.
Brooke, H.C.J., Hairless Mice, J. Hered., 1926, vol. 17, pp. 173-174.
Konyukhov, B.V., Biologicheskoe modelirovanie nasledstvennykh boleznei cheloveka (Biological Modeling of Human Hereditary Disorders), Moscow: Meditsyna, 1969.
Cachon-Gonzalez, M.B., Fenner, S., Coffin, J.M., et al., Structure and Expression of the hairless Gene of Mice, Proc. Natl. Acad. Sci. USA, 1994, vol. 91, pp. 7717-7721.
Cachon-Gonzalez, M.B., San-Jose, I., Cano, A., et al., The hairless Gene of the Mouse: Relationship of Phenotypic Effects with Expression Profile and Genotype, Dev. Dynam., 1999, vol. 216, pp. 113-126.
Panteleyev, A.A., Botchkareva, N.V., Sundberg, J.P., et al., The Role of the hairless (hr) Gene in the Regulation of Hair Follicle Catagen Transformation, Am. J. Pathol., 1999, vol. 155, pp. 159-171.
Panteleyev, A.A., Paus, R., and Christiano, A.M., Patterns of hairless(hr) Gene Expression in Mouse Hair Follicle Morphogenesis and Cycling, Am. J. Pathol., 2000, vol. 157, pp. 1071-1079.
David, L.T.J., Studies on the Expression of Genetic Hairlessness in the House Mouse (Mus musculus), J. Exp. Zool., 1934, vol. 68, pp. 501-518.
Konyukhov, B.V., Kupriyanov, S.D., and Isabekov, B.S., Use of Chimeric and Transgenic Animals to Study Gene Expression in Ontogeny, Usp. Sovrem. Genet., 1988, no. 15, pp. 106-142.
Mintz, B., Allophenic Mice of Multi-Embryo Origin, Methods in Mammalian Embryology, Deniel, J.C., Ed., San Francisco: Freeman, 1971, pp. 186-214.
Mintz, B., Gearhart, J.D., and Guymont, A.O., Phytohemagglutinin Mediated Blastomere Aggregation and Development of Allophenic Mice, Dev. Biol., 1973, vol. 31, pp. 195-199.
Whitten, W.K., Nutrient Requirements for the Culture of Preimplantation Embryos in Vitro, Adv. Biosci. (Oxford), 1971, vol. 6, pp. 129-141.
Abramczuk, J., Solter, D., and Koprowski, H., The Beneficial Effect of EDTA on Development of Mouse One-Cell Embryos in Chemically Defined Medium, Dev.Biol., 1977, vol. 61, pp. 378-383.
Brown, N.A., McCarthy, A., and Wolpert, L., The Development of Handed Asymmetry in Aggregation Chimeras of situs inversus Mutant and Wild-Type Mouse Embryo, Development (Cambridge, UK), 1990, vol. 110, pp. 949-954.
Isaev, D.A., Martynova, M.Yu., Platonov, E.S., and Konyukhov, B.V., Different Effect of Genome Imprinting on the Development of Parthenogenetic Cell Clones in C57BL/6 and CBA Mice, Ontogenez, 2001, vol. 32, no. 5, pp. 353-359.
Stenn, K.S. and Paus, R., Control of Hair Follicle Cycling, Physiol. Rev., 2001, vol. 81, pp. 449-494.
Potter, G.B. and Beaudoin, G.M.J. III, DeRenzo, C.L., et al., The hairless Gene Mutated in Congenital Hair Loss Disorders Encodes a Novel Nuclear Receptor Corepressor, Genes Dev., 2001, vol. 15, pp. 2687-2701.
Berdaliev, A.S. and Konyukhov, B.V., A Study of the Site of Action of the angora-Y Gene and Its Interaction with the fuzzy-Y Gene in Mouse, Izv. Akad. Nauk SSSR, Ser. Biol., 1991, no. 3, pp. 352-360.
Malinina, N.A., Martynova, M.Yu., and Konyukhov, B.V., The Mutant wal Gene Acts in Mouse Hair Follicle Cells, Ontogenez (Moscow), 1999, vol. 30, no. 5, pp. 362-365.
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Martynova, M.Y., Isaev, D.A. & Konyukhov, B.V. The Effects of Mutant Gene hairless in Chimeric Mice. Russian Journal of Genetics 39, 1056–1060 (2003). https://doi.org/10.1023/A:1025783303409
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DOI: https://doi.org/10.1023/A:1025783303409