A new allele of the mouse hairless gene interferes with Hox/LacZ transgene regulation in hair follicle primordia

Maud Virginie Brancaz, Rabah Iratni, Alastair Morrison, Stéphane J.C. Mancini, Patrice Marche, John Sundberg, Stefan Nonchev

Research output: Contribution to journalArticlepeer-review

15 Citations (Scopus)

Abstract

A new autosomal recessive mouse mutation, causing loss of hair in homozygous mice 2-3 weeks after birth, arose spontaneously in a colony at the National Institute for Medical Research (NIMR), Mill Hill, London in early 1998. Complementation analysis confirmed that this mutation was an allele of the hairless gene (hr). The gene symbol hrrhbm (hairless-rhino-bald Mill Hill) was assigned to reflect the source of the colony. Here we show the molecular defect in these mutants, which is a substantial deletion at the 3′-end of the hairless gene. Morphological and immunological analysis of the new hairless mutation was performed at early postnatal stages. In an effort to address the molecular and cellular mechanisms of the hairless phenotype, we analysed developmental stages before the establishment of alopecia. Using a HoxLacZ reporter line of transgenic mice, epidermal placode formation was followed in embryos. Homozygous mutant embryos (hrrhbmh/hr rhbmh), containing the LacZ reporter under the control of a Hoxb4 gene enhancer, display sharp loss of LacZ staining in epidermal cells invaginating to form the embryonic hair follicle placode. In the light of targeted mutagenesis data involving a Hox gene in the hair development [Genes Dev. 12 (1) (1998) 11], we discuss the potential implication of the hr rhbmh locus in cascades of Hox gene regulation during embryogenesis.

Original languageEnglish
Pages (from-to)173-181
Number of pages9
JournalExperimental and Molecular Pathology
Volume76
Issue number2
DOIs
Publication statusPublished - Apr 2004
Externally publishedYes

Keywords

  • Embryogenesis
  • Epidermis
  • Hair follicle
  • Hairless gene
  • Hairless mice
  • Hox gene regulation
  • Transgenic mice

ASJC Scopus subject areas

  • Pathology and Forensic Medicine
  • Molecular Biology
  • Clinical Biochemistry

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