Filaggrin expression and processing deficiencies impair corneocyte surface texture and stiffness in mice

Thyssen, Jacob P.; Jakasa, Ivone; Riethmüller, Christoph; Schön, Michael P.; Braun, Andrea; Haftek, Marek; Fallon, Padraic G.; Wróblewski, Jacek; Jakubowski, Hieronim; Eckhart, Leopold; Declercq, Wim; Koppes, Sjors; Engebretsen, Kristiane A.; Bonefeld, Charlotte; Irvine, Alan D.; Keita-Alassane, Sokhna; Simon, Michel; Kawasaki, Hiroshi; Kubo, Akiharu; Amagai, Masayuki; Matsui, Takeshi; Kezic, Sanja

Filaggrin expression and processing deficiencies impair corneocyte surface texture and stiffness in mice

Jacob P. Thyssen, Ivone Jakasa, Christoph Riethmüller, Michael P. Schön, Andrea Braun, Marek Haftek, Padraic G. Fallon, Jacek Wróblewski, Hieronim Jakubowski, Leopold Eckhart, et al.

(2020) JOURNAL OF INVESTIGATIVE DERMATOLOGY. 140(3). p.615-623.e5

Author

Jacob P. Thyssen, Ivone Jakasa, Christoph Riethmüller, Michael P. Schön, Andrea Braun, Marek Haftek, Padraic G. Fallon, Jacek Wróblewski, Hieronim Jakubowski, Leopold Eckhart, Wim Declercq (UGent) , Sjors Koppes, Kristiane A. Engebretsen, Charlotte Bonefeld, Alan D. Irvine, Sokhna Keita-Alassane, Michel Simon, Hiroshi Kawasaki, Akiharu Kubo, Masayuki Amagai, Takeshi Matsui and Sanja Kezic

Organization

Abstract

Abundant corneocyte surface protrusions, observed in patients with atopic dermatitis with filaggrin loss-of-function mutations, are inversely associated with levels of natural moisturizing factors (NMFs) in the stratum corneum. To dissect the etiological role of NMFs and filaggrin deficiency in surface texture alterations, we examined mouse models with genetic deficiencies in the synthesis or degradation of filaggrin monomers for NMFs, cell stiffness (elastic modulus) and corneocyte surface protrusion density (dermal texture index). Five neonatal and adult mouse models carrying inactivating mutations of SASPase (Sasp−/−), filaggrin (Flgft/ft and Flg−/−), filaggrin-hornerin (FlgHrnr−/−), and bleomycin hydrolase (Blmh−/−) were investigated. Sasp−/− and Flg−/− were on the hairless mouse background. Atomic force microscopy was used to determine elastic modulus and dermal texture index. Corneocytes of each neonatal as well as hairless adult knockout mouse exhibited an increased number of protrusions and decreased elastic modulus. In these mice, NMFs were reduced except for Sasp−/−. Dermal texture index was inversely correlated with NMFs and elastic modulus. Our findings demonstrate that any filaggrin-NMF axis deficiency can affect corneocyte mechanical properties in mice and likely in humans. Differences in NMFs and corneocyte surface texture between neonatal and adult as well as hairless and hairy mice emphasize the need for carefully selecting the most appropriate animal models for studies.

Keywords

Cell Biology, Biochemistry, Molecular Biology, Dermatology, PROTEASE BLEOMYCIN HYDROLASE, BARRIER, PROFILAGGRIN, BREAKDOWN, GENE, SKIN

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Please use this url to cite or link to this publication: http://hdl.handle.net/1854/LU-8650603

MLA: Thyssen, Jacob P., et al. “Filaggrin Expression and Processing Deficiencies Impair Corneocyte Surface Texture and Stiffness in Mice.” JOURNAL OF INVESTIGATIVE DERMATOLOGY, vol. 140, no. 3, 2020, pp. 615-623.e5, doi:10.1016/j.jid.2019.07.716.
APA: Thyssen, J. P., Jakasa, I., Riethmüller, C., Schön, M. P., Braun, A., Haftek, M., … Kezic, S. (2020). Filaggrin expression and processing deficiencies impair corneocyte surface texture and stiffness in mice. JOURNAL OF INVESTIGATIVE DERMATOLOGY, 140(3), 615-623.e5. https://doi.org/10.1016/j.jid.2019.07.716
Chicago author-date: Thyssen, Jacob P., Ivone Jakasa, Christoph Riethmüller, Michael P. Schön, Andrea Braun, Marek Haftek, Padraic G. Fallon, et al. 2020. “Filaggrin Expression and Processing Deficiencies Impair Corneocyte Surface Texture and Stiffness in Mice.” JOURNAL OF INVESTIGATIVE DERMATOLOGY 140 (3): 615-623.e5. https://doi.org/10.1016/j.jid.2019.07.716.
Chicago author-date (all authors): Thyssen, Jacob P., Ivone Jakasa, Christoph Riethmüller, Michael P. Schön, Andrea Braun, Marek Haftek, Padraic G. Fallon, Jacek Wróblewski, Hieronim Jakubowski, Leopold Eckhart, Wim Declercq, Sjors Koppes, Kristiane A. Engebretsen, Charlotte Bonefeld, Alan D. Irvine, Sokhna Keita-Alassane, Michel Simon, Hiroshi Kawasaki, Akiharu Kubo, Masayuki Amagai, Takeshi Matsui, and Sanja Kezic. 2020. “Filaggrin Expression and Processing Deficiencies Impair Corneocyte Surface Texture and Stiffness in Mice.” JOURNAL OF INVESTIGATIVE DERMATOLOGY 140 (3): 615-623.e5. doi:10.1016/j.jid.2019.07.716.
Vancouver: 1.
Thyssen JP, Jakasa I, Riethmüller C, Schön MP, Braun A, Haftek M, et al. Filaggrin expression and processing deficiencies impair corneocyte surface texture and stiffness in mice. JOURNAL OF INVESTIGATIVE DERMATOLOGY. 2020;140(3):615-623.e5.
IEEE: [1]
J. P. Thyssen et al., “Filaggrin expression and processing deficiencies impair corneocyte surface texture and stiffness in mice,” JOURNAL OF INVESTIGATIVE DERMATOLOGY, vol. 140, no. 3, pp. 615-623.e5, 2020.

@article{8650603,
  abstract     = {{Abundant corneocyte surface protrusions, observed in patients with atopic dermatitis with filaggrin loss-of-function mutations, are inversely associated with levels of natural moisturizing factors (NMFs) in the stratum corneum. To dissect the etiological role of NMFs and filaggrin deficiency in surface texture alterations, we examined mouse models with genetic deficiencies in the synthesis or degradation of filaggrin monomers for NMFs, cell stiffness (elastic modulus) and corneocyte surface protrusion density (dermal texture index). Five neonatal and adult mouse models carrying inactivating mutations of SASPase (Sasp−/−), filaggrin (Flgft/ft and Flg−/−), filaggrin-hornerin (FlgHrnr−/−), and bleomycin hydrolase (Blmh−/−) were investigated. Sasp−/− and Flg−/− were on the hairless mouse background. Atomic force microscopy was used to determine elastic modulus and dermal texture index. Corneocytes of each neonatal as well as hairless adult knockout mouse exhibited an increased number of protrusions and decreased elastic modulus. In these mice, NMFs were reduced except for Sasp−/−. Dermal texture index was inversely correlated with NMFs and elastic modulus. Our findings demonstrate that any filaggrin-NMF axis deficiency can affect corneocyte mechanical properties in mice and likely in humans. Differences in NMFs and corneocyte surface texture between neonatal and adult as well as hairless and hairy mice emphasize the need for carefully selecting the most appropriate animal models for studies.}},
  author       = {{Thyssen, Jacob P. and Jakasa, Ivone and Riethmüller, Christoph and Schön, Michael P. and Braun, Andrea and Haftek, Marek and Fallon, Padraic G. and Wróblewski, Jacek and Jakubowski, Hieronim and Eckhart, Leopold and Declercq, Wim and Koppes, Sjors and Engebretsen, Kristiane A. and Bonefeld, Charlotte and Irvine, Alan D. and Keita-Alassane, Sokhna and Simon, Michel and Kawasaki, Hiroshi and Kubo, Akiharu and Amagai, Masayuki and Matsui, Takeshi and Kezic, Sanja}},
  issn         = {{0022-202X}},
  journal      = {{JOURNAL OF INVESTIGATIVE DERMATOLOGY}},
  keywords     = {{Cell Biology,Biochemistry,Molecular Biology,Dermatology,PROTEASE BLEOMYCIN HYDROLASE,BARRIER,PROFILAGGRIN,BREAKDOWN,GENE,SKIN}},
  language     = {{eng}},
  number       = {{3}},
  pages        = {{615--623.e5}},
  title        = {{Filaggrin expression and processing deficiencies impair corneocyte surface texture and stiffness in mice}},
  url          = {{http://dx.doi.org/10.1016/j.jid.2019.07.716}},
  volume       = {{140}},
  year         = {{2020}},
}

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Filaggrin expression and processing deficiencies impair corneocyte surface texture and stiffness in mice

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