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Transcriptional profiling of rare acantholytic disorders suggests common mechanisms of pathogenesis

Darier, Hailey-Hailey, and Grover diseases are rare acantholytic skin diseases. While these diseases have different underlying causes, they share defects in cell-cell adhesion in the epidermis and desmosome organization. To better understand the underlying mechanisms leading to disease in these cond...

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Autores principales: Roth-Carter, Quinn R., Burks, Hope E., Ren, Ziyou, Koetsier, Jennifer L., Tsoi, Lam C., Harms, Paul W., Xing, Xianying, Kirma, Joseph, Harmon, Robert M., Godsel, Lisa M., Perl, Abbey L., Gudjonsson, Johann E., Green, Kathleen J.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: American Society for Clinical Investigation 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10543711/
https://www.ncbi.nlm.nih.gov/pubmed/37471166
http://dx.doi.org/10.1172/jci.insight.168955
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author Roth-Carter, Quinn R.
Burks, Hope E.
Ren, Ziyou
Koetsier, Jennifer L.
Tsoi, Lam C.
Harms, Paul W.
Xing, Xianying
Kirma, Joseph
Harmon, Robert M.
Godsel, Lisa M.
Perl, Abbey L.
Gudjonsson, Johann E.
Green, Kathleen J.
author_facet Roth-Carter, Quinn R.
Burks, Hope E.
Ren, Ziyou
Koetsier, Jennifer L.
Tsoi, Lam C.
Harms, Paul W.
Xing, Xianying
Kirma, Joseph
Harmon, Robert M.
Godsel, Lisa M.
Perl, Abbey L.
Gudjonsson, Johann E.
Green, Kathleen J.
author_sort Roth-Carter, Quinn R.
collection PubMed
description Darier, Hailey-Hailey, and Grover diseases are rare acantholytic skin diseases. While these diseases have different underlying causes, they share defects in cell-cell adhesion in the epidermis and desmosome organization. To better understand the underlying mechanisms leading to disease in these conditions, we performed RNA-seq on lesional skin samples from patients. The transcriptomic profiles of Darier, Hailey-Hailey, and Grover diseases were found to share a remarkable overlap, which did not extend to other common inflammatory skin diseases. Analysis of enriched pathways showed a shared increase in keratinocyte differentiation, and a decrease in cell adhesion and actin organization pathways in Darier, Hailey-Hailey, and Grover diseases. Direct comparison to atopic dermatitis and psoriasis showed that the downregulation in actin organization pathways was a unique feature in the acantholytic skin diseases. Furthermore, upstream regulator analysis suggested that a decrease in SRF/MRTF activity was responsible for the downregulation of actin organization pathways. Staining for MRTFA in lesional skin samples showed a decrease in nuclear MRTFA in patient skin compared with normal skin. These findings highlight the significant level of similarity in the transcriptome of Darier, Hailey-Hailey, and Grover diseases, and identify decreases in actin organization pathways as a unique signature present in these conditions.
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spelling pubmed-105437112023-10-03 Transcriptional profiling of rare acantholytic disorders suggests common mechanisms of pathogenesis Roth-Carter, Quinn R. Burks, Hope E. Ren, Ziyou Koetsier, Jennifer L. Tsoi, Lam C. Harms, Paul W. Xing, Xianying Kirma, Joseph Harmon, Robert M. Godsel, Lisa M. Perl, Abbey L. Gudjonsson, Johann E. Green, Kathleen J. JCI Insight Technical Advance Darier, Hailey-Hailey, and Grover diseases are rare acantholytic skin diseases. While these diseases have different underlying causes, they share defects in cell-cell adhesion in the epidermis and desmosome organization. To better understand the underlying mechanisms leading to disease in these conditions, we performed RNA-seq on lesional skin samples from patients. The transcriptomic profiles of Darier, Hailey-Hailey, and Grover diseases were found to share a remarkable overlap, which did not extend to other common inflammatory skin diseases. Analysis of enriched pathways showed a shared increase in keratinocyte differentiation, and a decrease in cell adhesion and actin organization pathways in Darier, Hailey-Hailey, and Grover diseases. Direct comparison to atopic dermatitis and psoriasis showed that the downregulation in actin organization pathways was a unique feature in the acantholytic skin diseases. Furthermore, upstream regulator analysis suggested that a decrease in SRF/MRTF activity was responsible for the downregulation of actin organization pathways. Staining for MRTFA in lesional skin samples showed a decrease in nuclear MRTFA in patient skin compared with normal skin. These findings highlight the significant level of similarity in the transcriptome of Darier, Hailey-Hailey, and Grover diseases, and identify decreases in actin organization pathways as a unique signature present in these conditions. American Society for Clinical Investigation 2023-08-22 /pmc/articles/PMC10543711/ /pubmed/37471166 http://dx.doi.org/10.1172/jci.insight.168955 Text en © 2023 Roth-Carter et al. https://creativecommons.org/licenses/by/4.0/This work is licensed under the Creative Commons Attribution 4.0 International License. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) .
spellingShingle Technical Advance
Roth-Carter, Quinn R.
Burks, Hope E.
Ren, Ziyou
Koetsier, Jennifer L.
Tsoi, Lam C.
Harms, Paul W.
Xing, Xianying
Kirma, Joseph
Harmon, Robert M.
Godsel, Lisa M.
Perl, Abbey L.
Gudjonsson, Johann E.
Green, Kathleen J.
Transcriptional profiling of rare acantholytic disorders suggests common mechanisms of pathogenesis
title Transcriptional profiling of rare acantholytic disorders suggests common mechanisms of pathogenesis
title_full Transcriptional profiling of rare acantholytic disorders suggests common mechanisms of pathogenesis
title_fullStr Transcriptional profiling of rare acantholytic disorders suggests common mechanisms of pathogenesis
title_full_unstemmed Transcriptional profiling of rare acantholytic disorders suggests common mechanisms of pathogenesis
title_short Transcriptional profiling of rare acantholytic disorders suggests common mechanisms of pathogenesis
title_sort transcriptional profiling of rare acantholytic disorders suggests common mechanisms of pathogenesis
topic Technical Advance
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10543711/
https://www.ncbi.nlm.nih.gov/pubmed/37471166
http://dx.doi.org/10.1172/jci.insight.168955
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