Desmoglein 4 in hair follicle differentiation and epidermal adhesion: Evidence from inherited hypotrichosis and acquired pemphigus vulgaris

Ana Kljuic; Hisham Bazzi; John P. Sundberg; Amalia Martinez-Mir; Ryan O'Shaughnessy; My G. Mahoney; Moise Levy; Xavier Montagutelli; Wasim Ahmad; Vincent M. Aita; Derek Gordon; Jouni Uitto; David Whiting; Jurg Ott; Stuart Fischer; T. Conrad Gilliam; Colin A B Jahoda; Rebecca J. Morris; Andrei A. Panteleyev; Vu Thuong Nguyen; Angela M. Christiano

doi:10.1016/S0092-8674(03)00273-3

Desmoglein 4 in hair follicle differentiation and epidermal adhesion: Evidence from inherited hypotrichosis and acquired pemphigus vulgaris

Ana Kljuic, Hisham Bazzi, John P. Sundberg, Amalia Martinez-Mir, Ryan O'Shaughnessy, My G. Mahoney, Moise Levy, Xavier Montagutelli, Wasim Ahmad, Vincent M. Aita, Derek Gordon, Jouni Uitto, David Whiting, Jurg Ott, Stuart Fischer, T. Conrad Gilliam, Colin A B Jahoda, Rebecca J. Morris, Andrei A. Panteleyev, Vu Thuong NguyenAngela M. Christiano

The Hormel Institute

Research output: Contribution to journal › Article › peer-review

295 Scopus citations

Abstract

Cell adhesion and communication are interdependent aspects of cell behavior that are critical for morphogenesis and tissue architecture. In the skin, epidermal adhesion is mediated in part by specialized cell-cell junctions known as desmosomes, which are characterized by the presence of desmosomal cadherins, known as desmogleins and desmocollins. We identified a cadherin family member, desmoglein 4, which is expressed in the suprabasal epidermis and hair follicle. The essential role of desmoglein 4 in skin was established by identifying mutations in families with inherited hypotrichosis, as well as in the lanceolate hair mouse. We also show that DSG4 is an autoantigen in pemphigus vulgaris. Characterization of the phenotype of naturally occurring mutant mice revealed disruption of desmosomal adhesion and perturbations in keratinocyte behavior. We provide evidence that desmoglein 4 is a key mediator of keratinocyte cell adhesion in the hair follicle, where it coordinates the transition from proliferation to differentiation.

Original language	English (US)
Pages (from-to)	249-260
Number of pages	12
Journal	Cell
Volume	113
Issue number	2
DOIs	https://doi.org/10.1016/S0092-8674(03)00273-3
State	Published - Apr 18 2003

Bibliographical note

Funding Information:
We thank the family members for their interest and participation in this work. We appreciate discussions with Drs. David Owens, Ronald Liem, Akemi Ishida-Yamamoto, John Mc Grath, Abraham Zlotogorski, Howard Baden, Lloyd King, Elise Olsen, Lowell Goldsmith, and Cathy Mendelsohn. We thank Drs. Gary Struhl and Andreu Casali-Taberner for their generous help with confocal microscopy. We thank Andrew Engelhard, Karima Djabali, Marija Tadin, Hyunmi Kim, Kai Sun, Ming Zhang, Nancy Zhou, and HaMut Lam. This work was supported in part by USPHS NIH R01-44924 (AMC), K26-RR173 (JPS), P01 AR38923 (JU), R03 AR47938 (MGM), and the March of Dimes Birth Defects Foundation (AMC).

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Kljuic, A., Bazzi, H., Sundberg, J. P., Martinez-Mir, A., O'Shaughnessy, R., Mahoney, M. G., Levy, M., Montagutelli, X., Ahmad, W., Aita, V. M., Gordon, D., Uitto, J., Whiting, D., Ott, J., Fischer, S., Gilliam, T. C., Jahoda, C. A. B., Morris, R. J., Panteleyev, A. A., ... Christiano, A. M. (2003). Desmoglein 4 in hair follicle differentiation and epidermal adhesion: Evidence from inherited hypotrichosis and acquired pemphigus vulgaris. Cell, 113(2), 249-260. https://doi.org/10.1016/S0092-8674(03)00273-3

Kljuic, A, Bazzi, H, Sundberg, JP, Martinez-Mir, A, O'Shaughnessy, R, Mahoney, MG, Levy, M, Montagutelli, X, Ahmad, W, Aita, VM, Gordon, D, Uitto, J, Whiting, D, Ott, J, Fischer, S, Gilliam, TC, Jahoda, CAB, Morris, RJ, Panteleyev, AA, Nguyen, VT & Christiano, AM 2003, 'Desmoglein 4 in hair follicle differentiation and epidermal adhesion: Evidence from inherited hypotrichosis and acquired pemphigus vulgaris', Cell, vol. 113, no. 2, pp. 249-260. https://doi.org/10.1016/S0092-8674(03)00273-3

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title = "Desmoglein 4 in hair follicle differentiation and epidermal adhesion: Evidence from inherited hypotrichosis and acquired pemphigus vulgaris",

abstract = "Cell adhesion and communication are interdependent aspects of cell behavior that are critical for morphogenesis and tissue architecture. In the skin, epidermal adhesion is mediated in part by specialized cell-cell junctions known as desmosomes, which are characterized by the presence of desmosomal cadherins, known as desmogleins and desmocollins. We identified a cadherin family member, desmoglein 4, which is expressed in the suprabasal epidermis and hair follicle. The essential role of desmoglein 4 in skin was established by identifying mutations in families with inherited hypotrichosis, as well as in the lanceolate hair mouse. We also show that DSG4 is an autoantigen in pemphigus vulgaris. Characterization of the phenotype of naturally occurring mutant mice revealed disruption of desmosomal adhesion and perturbations in keratinocyte behavior. We provide evidence that desmoglein 4 is a key mediator of keratinocyte cell adhesion in the hair follicle, where it coordinates the transition from proliferation to differentiation.",

author = "Ana Kljuic and Hisham Bazzi and Sundberg, {John P.} and Amalia Martinez-Mir and Ryan O'Shaughnessy and Mahoney, {My G.} and Moise Levy and Xavier Montagutelli and Wasim Ahmad and Aita, {Vincent M.} and Derek Gordon and Jouni Uitto and David Whiting and Jurg Ott and Stuart Fischer and Gilliam, {T. Conrad} and Jahoda, {Colin A B} and Morris, {Rebecca J.} and Panteleyev, {Andrei A.} and Nguyen, {Vu Thuong} and Christiano, {Angela M.}",

note = "Funding Information: We thank the family members for their interest and participation in this work. We appreciate discussions with Drs. David Owens, Ronald Liem, Akemi Ishida-Yamamoto, John Mc Grath, Abraham Zlotogorski, Howard Baden, Lloyd King, Elise Olsen, Lowell Goldsmith, and Cathy Mendelsohn. We thank Drs. Gary Struhl and Andreu Casali-Taberner for their generous help with confocal microscopy. We thank Andrew Engelhard, Karima Djabali, Marija Tadin, Hyunmi Kim, Kai Sun, Ming Zhang, Nancy Zhou, and HaMut Lam. This work was supported in part by USPHS NIH R01-44924 (AMC), K26-RR173 (JPS), P01 AR38923 (JU), R03 AR47938 (MGM), and the March of Dimes Birth Defects Foundation (AMC). ",

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doi = "10.1016/S0092-8674(03)00273-3",

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T1 - Desmoglein 4 in hair follicle differentiation and epidermal adhesion

T2 - Evidence from inherited hypotrichosis and acquired pemphigus vulgaris

AU - Kljuic, Ana

AU - Bazzi, Hisham

AU - Sundberg, John P.

AU - Martinez-Mir, Amalia

AU - O'Shaughnessy, Ryan

AU - Mahoney, My G.

AU - Levy, Moise

AU - Montagutelli, Xavier

AU - Ahmad, Wasim

AU - Aita, Vincent M.

AU - Gordon, Derek

AU - Uitto, Jouni

AU - Whiting, David

AU - Ott, Jurg

AU - Fischer, Stuart

AU - Gilliam, T. Conrad

AU - Jahoda, Colin A B

AU - Morris, Rebecca J.

AU - Panteleyev, Andrei A.

AU - Nguyen, Vu Thuong

AU - Christiano, Angela M.

N1 - Funding Information: We thank the family members for their interest and participation in this work. We appreciate discussions with Drs. David Owens, Ronald Liem, Akemi Ishida-Yamamoto, John Mc Grath, Abraham Zlotogorski, Howard Baden, Lloyd King, Elise Olsen, Lowell Goldsmith, and Cathy Mendelsohn. We thank Drs. Gary Struhl and Andreu Casali-Taberner for their generous help with confocal microscopy. We thank Andrew Engelhard, Karima Djabali, Marija Tadin, Hyunmi Kim, Kai Sun, Ming Zhang, Nancy Zhou, and HaMut Lam. This work was supported in part by USPHS NIH R01-44924 (AMC), K26-RR173 (JPS), P01 AR38923 (JU), R03 AR47938 (MGM), and the March of Dimes Birth Defects Foundation (AMC).

PY - 2003/4/18

Y1 - 2003/4/18

N2 - Cell adhesion and communication are interdependent aspects of cell behavior that are critical for morphogenesis and tissue architecture. In the skin, epidermal adhesion is mediated in part by specialized cell-cell junctions known as desmosomes, which are characterized by the presence of desmosomal cadherins, known as desmogleins and desmocollins. We identified a cadherin family member, desmoglein 4, which is expressed in the suprabasal epidermis and hair follicle. The essential role of desmoglein 4 in skin was established by identifying mutations in families with inherited hypotrichosis, as well as in the lanceolate hair mouse. We also show that DSG4 is an autoantigen in pemphigus vulgaris. Characterization of the phenotype of naturally occurring mutant mice revealed disruption of desmosomal adhesion and perturbations in keratinocyte behavior. We provide evidence that desmoglein 4 is a key mediator of keratinocyte cell adhesion in the hair follicle, where it coordinates the transition from proliferation to differentiation.

AB - Cell adhesion and communication are interdependent aspects of cell behavior that are critical for morphogenesis and tissue architecture. In the skin, epidermal adhesion is mediated in part by specialized cell-cell junctions known as desmosomes, which are characterized by the presence of desmosomal cadherins, known as desmogleins and desmocollins. We identified a cadherin family member, desmoglein 4, which is expressed in the suprabasal epidermis and hair follicle. The essential role of desmoglein 4 in skin was established by identifying mutations in families with inherited hypotrichosis, as well as in the lanceolate hair mouse. We also show that DSG4 is an autoantigen in pemphigus vulgaris. Characterization of the phenotype of naturally occurring mutant mice revealed disruption of desmosomal adhesion and perturbations in keratinocyte behavior. We provide evidence that desmoglein 4 is a key mediator of keratinocyte cell adhesion in the hair follicle, where it coordinates the transition from proliferation to differentiation.

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Desmoglein 4 in hair follicle differentiation and epidermal adhesion: Evidence from inherited hypotrichosis and acquired pemphigus vulgaris

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