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Cited by 22Hair Growth and DisordersSkin and Cellular Biology Researchmelanin and skin pigmentation

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Progressive Hair Loss and Myocardial Degeneration in Rough Coat Mice: Reduced Lysyl Oxidase-Like (LOXL) in the Skin and Heart

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Hayashi, Kimiko, Cao, Tongyu, Passmore, Howard, Saux, Claude Jourdan-Le, Fogelgren, Ben, Khan, Subarna, Hornstra, Ian, Kim, Youngho, Hayashi, Masando, Csiszar, Katalin·2004·Journal of Investigative Dermatology
DOI 10.1111/j.0022-202x.2004.23436.x
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Abstract

The rough coat (rc) is a spontaneous recessive mutation in mice. To identify the mutated gene, we have characterized the rc phenotype and initiated linkage mapping. The rc mice show growth retardation, cyclic and progressive hair loss, hyperplastic epidermis, abnormal hair follicles, cardiac muscle degeneration, and reduced amount of collagen and elastin in the skin and heart. The rc locus was mapped at 32.0 cM on chromosome 9, close to the loxl gene. Lysyl oxidase-like (LOXL) protein is a novel copper-containing amine oxidase that is required for the cross-linking of elastin and collagen in vitro. LOXL is expressed at high levels in the skin and heart, where the rc mice show strong phenotype. The expression pattern and the genetic proximity to rc suggested loxl as a potential candidate gene. In rc mice, the loxl mRNA was reduced in the skin and the LOXL protein in the heart, dermis, atrophic hair follicles, and sebaceous glands. No mutations, however, were identified within the coding region of loxl, and offspring from rc/rc and loxl null mice crossing were phenotypically normal. Based on these results, loxl appears non-allelic to rc. Heart- and skin-specific downregulation of LOXL in rc mice, however, may contribute to the extracellular matrix alterations and the rc phenotype.

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Cited by

Metadata

Authors
Hayashi, K., Cao, T., Passmore, H., Saux, C. J.-L., Fogelgren, B., Khan, S., Hornstra, I., Kim, Y., Hayashi, M., & Csiszar, K.
Affiliations
  • University of Hawaiʻi at Mānoa (US)
  • Rutgers, The State University of New Jersey (US)
  • Barnes-Jewish Hospital (US)
  • Asan Medical Center (KR)
ORCID
Venue
Journal of Investigative Dermatology
Publisher
Elsevier BV
Published
2004-11
Volume
123
Issue
5
Pages
864-871
ISSN
0022-202X (ISSN-L)
DOI
10.1111/j.0022-202x.2004.23436.x
OpenAlex
W2002447427
Cited by
22
References
0
Citations by year
2023: 1 · 2021: 1 · 2020: 1 · 2017: 2 · 2016: 1 · 2015: 2 · 2014: 1 · 2013: 2 · 2012: 2
FWCI
1.00
Type
journal article
Language
en
Open access
Licence
Locations
  • Journal of Investigative Dermatology (publishedVersion)
  • PubMed (publishedVersion)
Funders
  • National Institutes of Health (5r29ca076580-05)
  • National Cancer Institute (CA76580)
  • National Institute of Arthritis and Musculoskeletal and Skin Diseases (S11AR047713, AR47713)
  • National Center for Research Resources (RR16453, P20RR016453, RR03061)
Topics
  • Health Sciences › Medicine › Dermatology › Hair Growth and Disorders
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › Skin and Cellular Biology Research
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › melanin and skin pigmentation
Keywords
hair loss, myocardial degeneration, collagen cross-linking, elastin, extracellular matrix, epidermal hyperplasia, hair follicle, sebaceous glands, linkage mapping, recessive mutations
MeSH terms
Alopecia, Amino Acid Oxidoreductases, Animals, Collagen, Elastin, Epidermis, Female, Immunohistochemistry, Genetic Linkage, Male, Mice, Inbred BALB C, Mice, Inbred C57BL, Mice, Mutant Strains, Myocardium, Phenotype, RNA, Messenger, Mice

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