The near-naked hairless (HrN) mutation disrupts hair formation but is not due to a mutation in the hairless coding region

Yutao Liu, Suchita Das, Robert E. Olszewski, Donald A. Carpenter, Cymbeline T. Culiat, John P. Sundberg, Patricia Soteropoulos, Xiaochen Liu, Mitchel J. Doktycz, Edward J. Michaud, Brynn H. Voy

Research output: Contribution to journalArticle

9 Citations (Scopus)

Abstract

Near-naked hairless (HrN) is a semi-dominant, spontaneous mutation that was suggested by allelism testing to be allelic with mouse Hairless (Hr). HrN mice differ from other Hr mutants in that hair loss appears as the postnatal coat begins to emerge, rather than as an inability to regrow hair after the first catagen and that the mutation displays semi-dominant inheritance. We sequenced the Hr cDNA in HrN/Hr N mice and characterized the pathological and molecular phenotypes to identify the basis for hair loss in this model. HrN/HrN mice exhibit dystrophic hairs that are unable to emerge consistently from the hair follicle, whereas HrN/+ mice display a sparse coat of hair and a milder degree of follicular dystrophy than their homozygous littermates. DNA microarray analysis of cutaneous gene expression demonstrates that numerous genes are downregulated in HrN/HrN mice, primarily genes important for hair structure. By contrast, Hr expression is significantly increased. Sequencing the Hr-coding region, intron-exon boundaries, 5′- and 3′-untranslated region, and immediate upstream region did not reveal the underlying mutation. Therefore, HrN does not appear to be an allele of Hr but may result from a mutation in a closely linked gene or from a regulatory mutation in Hr.

Original languageEnglish (US)
Pages (from-to)1605-1614
Number of pages10
JournalJournal of Investigative Dermatology
Volume127
Issue number7
DOIs
StatePublished - Jul 1 2007

Fingerprint

Hair
Genes
Mutation
Hairless Mouse
Alopecia
5' Untranslated Regions
3' Untranslated Regions
Microarrays
Gene expression
Introns
Exons
Complementary DNA
Hair Follicle
Microarray Analysis
Oligonucleotide Array Sequence Analysis
DNA
Testing
Down-Regulation
Alleles
Phenotype

ASJC Scopus subject areas

  • Biochemistry
  • Molecular Biology
  • Dermatology
  • Cell Biology

Cite this

The near-naked hairless (HrN) mutation disrupts hair formation but is not due to a mutation in the hairless coding region. / Liu, Yutao; Das, Suchita; Olszewski, Robert E.; Carpenter, Donald A.; Culiat, Cymbeline T.; Sundberg, John P.; Soteropoulos, Patricia; Liu, Xiaochen; Doktycz, Mitchel J.; Michaud, Edward J.; Voy, Brynn H.

In: Journal of Investigative Dermatology, Vol. 127, No. 7, 01.07.2007, p. 1605-1614.

Research output: Contribution to journalArticle

Liu, Y, Das, S, Olszewski, RE, Carpenter, DA, Culiat, CT, Sundberg, JP, Soteropoulos, P, Liu, X, Doktycz, MJ, Michaud, EJ & Voy, BH 2007, 'The near-naked hairless (HrN) mutation disrupts hair formation but is not due to a mutation in the hairless coding region', Journal of Investigative Dermatology, vol. 127, no. 7, pp. 1605-1614. https://doi.org/10.1038/sj.jid.5700755
Liu, Yutao ; Das, Suchita ; Olszewski, Robert E. ; Carpenter, Donald A. ; Culiat, Cymbeline T. ; Sundberg, John P. ; Soteropoulos, Patricia ; Liu, Xiaochen ; Doktycz, Mitchel J. ; Michaud, Edward J. ; Voy, Brynn H. / The near-naked hairless (HrN) mutation disrupts hair formation but is not due to a mutation in the hairless coding region. In: Journal of Investigative Dermatology. 2007 ; Vol. 127, No. 7. pp. 1605-1614.
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abstract = "Near-naked hairless (HrN) is a semi-dominant, spontaneous mutation that was suggested by allelism testing to be allelic with mouse Hairless (Hr). HrN mice differ from other Hr mutants in that hair loss appears as the postnatal coat begins to emerge, rather than as an inability to regrow hair after the first catagen and that the mutation displays semi-dominant inheritance. We sequenced the Hr cDNA in HrN/Hr N mice and characterized the pathological and molecular phenotypes to identify the basis for hair loss in this model. HrN/HrN mice exhibit dystrophic hairs that are unable to emerge consistently from the hair follicle, whereas HrN/+ mice display a sparse coat of hair and a milder degree of follicular dystrophy than their homozygous littermates. DNA microarray analysis of cutaneous gene expression demonstrates that numerous genes are downregulated in HrN/HrN mice, primarily genes important for hair structure. By contrast, Hr expression is significantly increased. Sequencing the Hr-coding region, intron-exon boundaries, 5′- and 3′-untranslated region, and immediate upstream region did not reveal the underlying mutation. Therefore, HrN does not appear to be an allele of Hr but may result from a mutation in a closely linked gene or from a regulatory mutation in Hr.",
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