Progressive hair loss and myocardial degeneration in rough coat mice: Reduced lysyl oxidase-like (LOXL) in the skin and heart

Kimiko Hayashi, Tongyu Wikramanayake, Howard Passmore, Claude Jourdan-Le Saux, Ben Fogelgren, Subarna Khan, Ian Hornstra, Youngho Kim, Masando Hayashi, Katalin Csiszar

Research output: Contribution to journalArticle

13 Citations (Scopus)

Abstract

The rough coat (re) is a spontaneous recessive mutation in mice. To identify the mutated gene, we have characterized the re phenotype and initiated linkage mapping. The re 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 re mice show strong phenotype. The expression pattern and the genetic proximity to rc suggested loxl as a potential candidate gene. In re 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 re/re 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 re mice, however, may contribute to the extracellular matrix alterations and the re phenotype.

Original languageEnglish
Pages (from-to)864-871
Number of pages8
JournalJournal of Investigative Dermatology
Volume123
Issue number5
DOIs
StatePublished - Nov 1 2004
Externally publishedYes

Fingerprint

Protein-Lysine 6-Oxidase
Alopecia
Skin
Elastin
Genes
Collagen
Hair Follicle
Amine Oxidase (Copper-Containing)
Phenotype
Chromosomes
Muscle
Sebaceous Glands
Chromosomes, Human, Pair 9
Mutation
Proteins
Chromosome Mapping
Dermis
Epidermis
Messenger RNA
Extracellular Matrix

Keywords

  • Amine oxidase
  • Collagen
  • Elastin
  • Hair follicle
  • Sebaceous gland

ASJC Scopus subject areas

  • Dermatology

Cite this

Progressive hair loss and myocardial degeneration in rough coat mice : Reduced lysyl oxidase-like (LOXL) in the skin and heart. / Hayashi, Kimiko; Wikramanayake, Tongyu; Passmore, Howard; Jourdan-Le Saux, Claude; Fogelgren, Ben; Khan, Subarna; Hornstra, Ian; Kim, Youngho; Hayashi, Masando; Csiszar, Katalin.

In: Journal of Investigative Dermatology, Vol. 123, No. 5, 01.11.2004, p. 864-871.

Research output: Contribution to journalArticle

Hayashi, K, Wikramanayake, T, Passmore, H, Jourdan-Le Saux, C, Fogelgren, B, Khan, S, Hornstra, I, Kim, Y, Hayashi, M & Csiszar, K 2004, 'Progressive hair loss and myocardial degeneration in rough coat mice: Reduced lysyl oxidase-like (LOXL) in the skin and heart', Journal of Investigative Dermatology, vol. 123, no. 5, pp. 864-871. https://doi.org/10.1111/j.0022-202X.2004.23436.x
Hayashi, Kimiko ; Wikramanayake, Tongyu ; Passmore, Howard ; Jourdan-Le Saux, Claude ; Fogelgren, Ben ; Khan, Subarna ; Hornstra, Ian ; Kim, Youngho ; Hayashi, Masando ; Csiszar, Katalin. / Progressive hair loss and myocardial degeneration in rough coat mice : Reduced lysyl oxidase-like (LOXL) in the skin and heart. In: Journal of Investigative Dermatology. 2004 ; Vol. 123, No. 5. pp. 864-871.
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