Mouse models for spontaneous dermatitis due to impaired skin barrier formation

Research output: Contribution to journalArticle

1 Citation (Scopus)

Abstract

Flaky tail mice possess two distinct autosomal recessive mutations, hair abnormality (matted: ma) and stratum corneum layer abnormality (flaky tail: ft), and develop dermatitis spontaneously with high serum IgE even under specific pathogen free condition. We demonstrated that flaky tail mice possess loss of function mutation in Filaggrin (Flg) which is one of the major component of stratum corneum, and showed skin barrier abnormality, although our genetically engineered Flg null mice did not develop dermatitis spontaneously. As a result of segregation of ft and ma mutations, we identified that ma mutation is responsible for the dermatitis phenotype in flaky tail mice, and Tmem79 nonsense mutation (Tmem79<sup>ma</sup>) is corresponding to the ma mutation. Tmem79 is expressed in outermost cell of stratum granulosum layer, and ma mice showed abnormal lamellar granule secretory system and abnormal formation of stratum corneum. Thus, Tmem79<sup>ma/ma</sup> mice provides a useful mouse model for spontaneous dermatitis caused by skin barrier gene deficiency. Further analysis for Tmem79<sup>ma/ma</sup> mice would elucidate important mechanisms for development of atopic dermatitis.

Original languageEnglish
Pages (from-to)160-165
Number of pages6
JournalJapanese Journal of Clinical Immunology
Volume37
Issue number3
DOIs
Publication statusPublished - 2014

Fingerprint

Dermatitis
Skin
Tail
Nonsense Codon
Mutation
Cornea
Skin Abnormalities
Specific Pathogen-Free Organisms
Granulosa Cells
Secretory Vesicles
Atopic Dermatitis
Hair
Immunoglobulin E
Phenotype
Serum
Genes

Keywords

  • Atopic dermatitis
  • Flaggrin
  • Mouse model
  • Skin barrier
  • Spontaneous dermatitis

ASJC Scopus subject areas

  • Immunology and Allergy
  • Immunology
  • Medicine(all)

Cite this

Mouse models for spontaneous dermatitis due to impaired skin barrier formation. / Sasaki, Takashi; Shiohama, Aiko; Amagai, Masayuki.

In: Japanese Journal of Clinical Immunology, Vol. 37, No. 3, 2014, p. 160-165.

Research output: Contribution to journalArticle

@article{9af6b22b2b1e40398c3d97de24b07e9f,
title = "Mouse models for spontaneous dermatitis due to impaired skin barrier formation",
abstract = "Flaky tail mice possess two distinct autosomal recessive mutations, hair abnormality (matted: ma) and stratum corneum layer abnormality (flaky tail: ft), and develop dermatitis spontaneously with high serum IgE even under specific pathogen free condition. We demonstrated that flaky tail mice possess loss of function mutation in Filaggrin (Flg) which is one of the major component of stratum corneum, and showed skin barrier abnormality, although our genetically engineered Flg null mice did not develop dermatitis spontaneously. As a result of segregation of ft and ma mutations, we identified that ma mutation is responsible for the dermatitis phenotype in flaky tail mice, and Tmem79 nonsense mutation (Tmem79ma) is corresponding to the ma mutation. Tmem79 is expressed in outermost cell of stratum granulosum layer, and ma mice showed abnormal lamellar granule secretory system and abnormal formation of stratum corneum. Thus, Tmem79ma/ma mice provides a useful mouse model for spontaneous dermatitis caused by skin barrier gene deficiency. Further analysis for Tmem79ma/ma mice would elucidate important mechanisms for development of atopic dermatitis.",
keywords = "Atopic dermatitis, Flaggrin, Mouse model, Skin barrier, Spontaneous dermatitis",
author = "Takashi Sasaki and Aiko Shiohama and Masayuki Amagai",
year = "2014",
doi = "10.2177/jsci.37.160",
language = "English",
volume = "37",
pages = "160--165",
journal = "Immunological Medicine",
issn = "0911-4300",
publisher = "Taylor and Francis Ltd.",
number = "3",

}

TY - JOUR

T1 - Mouse models for spontaneous dermatitis due to impaired skin barrier formation

AU - Sasaki, Takashi

AU - Shiohama, Aiko

AU - Amagai, Masayuki

PY - 2014

Y1 - 2014

N2 - Flaky tail mice possess two distinct autosomal recessive mutations, hair abnormality (matted: ma) and stratum corneum layer abnormality (flaky tail: ft), and develop dermatitis spontaneously with high serum IgE even under specific pathogen free condition. We demonstrated that flaky tail mice possess loss of function mutation in Filaggrin (Flg) which is one of the major component of stratum corneum, and showed skin barrier abnormality, although our genetically engineered Flg null mice did not develop dermatitis spontaneously. As a result of segregation of ft and ma mutations, we identified that ma mutation is responsible for the dermatitis phenotype in flaky tail mice, and Tmem79 nonsense mutation (Tmem79ma) is corresponding to the ma mutation. Tmem79 is expressed in outermost cell of stratum granulosum layer, and ma mice showed abnormal lamellar granule secretory system and abnormal formation of stratum corneum. Thus, Tmem79ma/ma mice provides a useful mouse model for spontaneous dermatitis caused by skin barrier gene deficiency. Further analysis for Tmem79ma/ma mice would elucidate important mechanisms for development of atopic dermatitis.

AB - Flaky tail mice possess two distinct autosomal recessive mutations, hair abnormality (matted: ma) and stratum corneum layer abnormality (flaky tail: ft), and develop dermatitis spontaneously with high serum IgE even under specific pathogen free condition. We demonstrated that flaky tail mice possess loss of function mutation in Filaggrin (Flg) which is one of the major component of stratum corneum, and showed skin barrier abnormality, although our genetically engineered Flg null mice did not develop dermatitis spontaneously. As a result of segregation of ft and ma mutations, we identified that ma mutation is responsible for the dermatitis phenotype in flaky tail mice, and Tmem79 nonsense mutation (Tmem79ma) is corresponding to the ma mutation. Tmem79 is expressed in outermost cell of stratum granulosum layer, and ma mice showed abnormal lamellar granule secretory system and abnormal formation of stratum corneum. Thus, Tmem79ma/ma mice provides a useful mouse model for spontaneous dermatitis caused by skin barrier gene deficiency. Further analysis for Tmem79ma/ma mice would elucidate important mechanisms for development of atopic dermatitis.

KW - Atopic dermatitis

KW - Flaggrin

KW - Mouse model

KW - Skin barrier

KW - Spontaneous dermatitis

UR - http://www.scopus.com/inward/record.url?scp=84936753490&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=84936753490&partnerID=8YFLogxK

U2 - 10.2177/jsci.37.160

DO - 10.2177/jsci.37.160

M3 - Article

VL - 37

SP - 160

EP - 165

JO - Immunological Medicine

JF - Immunological Medicine

SN - 0911-4300

IS - 3

ER -