The hairless phenotype of the Hirosaki hairless rat is due to the deletion of an 80-kb genomic DNA containing five basic keratin genes

The Journal of Biological Chemistry
Naoki NanashimaShigeki Tsuchida

Abstract

Most models of hereditary hypotrichosis are due to alterations in growth factors and transcription factors, and the examples of causative mutations in hair keratin genes are limited. The Hirosaki hairless rat (HHR) is a mutant strain spontaneously derived from Sprague-Dawley rats (SDRs). In this study, the locus of the responsible gene was examined by linkage analysis and mapped on chromosome 7q36. Because many basic keratin genes are clustered on 7q36, their expression was examined. Reverse transcription-PCR and genomic PCR indicated that the Kb21 (Krt81), -23 (Krt83), and -26 (Krt86) genes encoding basic hair keratins were not expressed and were deleted. Furthermore, 80-kb genomic DNA ranging from exon 9 of Kb25 (Krt85) to exon 9 of Krt2-25 was deleted. The breakpoints of these genes were within a 95-bp portion shared by the two genes, suggesting that deletion due to non-allelic homologous recombination occurred. Proteins identified as Kb21, Kb23, and Krt2-25 in SDR hairs by mass spectrometry were not detected in HHR. Instead, the product of a fusion gene became dominant in HHR. Because fusion occurred between the exons of the two genes with the same sequences, the product was identical to the wild-type Kb25 protein. By using...Continue Reading

References

Jul 27, 1978·Nature·M F FestingS Sparrow
Sep 1, 1979·Proceedings of the National Academy of Sciences of the United States of America·H TowbinJ Gordon
Sep 1, 1976·The Journal of Heredity·J Palm, F G Ferguson
Jun 1, 1988·The Journal of Dermatology·K HanadaI Hatayama
Apr 1, 1981·The Journal of Histochemistry and Cytochemistry : Official Journal of the Histochemistry Society·S M HsuH Fanger
Feb 1, 1995·Current Opinion in Cell Biology·W H McLean, E B Lane
Feb 21, 1998·Genes & Development·A R Godwin, M R Capecchi
Jul 10, 1998·The Journal of Heredity·A N MoemekaT R King
Dec 18, 1998·The Journal of Investigative Dermatology·H WinterJ Schweizer
Mar 2, 1999·Developmental Biology·S E MillarG S Barsh
Jul 3, 1999·The Journal of Biological Chemistry·L LangbeinJ Schweizer
Jul 6, 2000·Developmental Dynamics : an Official Publication of the American Association of Anatomists·M SchorppT Boehm
Sep 5, 2002·Molecular Genetics and Metabolism·R R ChrissluisT R King
Oct 26, 2002·Genetics·Christophe PoirierMoriaki Kusakabe
Dec 17, 2003·The Journal of Investigative Dermatology·Zhiling WangPierre A Coulombe
Apr 17, 2004·European Journal of Cell Biology·Michael HesseThomas M Magin
Mar 31, 2005·International Review of Cytology·Lutz Langbein, Jürgen Schweizer
May 17, 2006·Genes & Development·Xuemei Tong, Pierre A Coulombe
Jul 13, 2006·The Journal of Cell Biology·Jürgen SchweizerMathew W Wright
Aug 31, 2006·Nature Genetics·James R Lupski

❮ Previous
Next ❯

Citations

Feb 3, 2011·Journal of Cell Science·Andrew H JheonOphir D Klein
Dec 7, 2014·Chinese Journal of Natural Medicines·Chun-Hua MaShi-Ping Ma
Oct 8, 2014·BMC Genomics·Mariana F NeryJuan C Opazo
Mar 24, 2017·Journal of Molecular Evolution·Naoki NanashimaShigeki Tsuchida
Feb 8, 2020·BMC Developmental Biology·He ZhangLingling Jin
Sep 3, 2013·The Journal of Immunology : Official Journal of the American Association of Immunologists·Toshiyuki YamadaShigeki Tsuchida

❮ Previous
Next ❯

Related Concepts

Related Feeds

ASBMB Publications

The American Society for Biochemistry and Molecular Biology (ASBMB) includes the Journal of Biological Chemistry, Molecular & Cellular Proteomics, and the Journal of Lipid Research. Discover the latest research from ASBMB here.