Cargando…
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...
Autores principales: | , , , , , , , , , , , , |
---|---|
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 |
_version_ | 1785114341457002496 |
---|---|
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. |
format | Online Article Text |
id | pubmed-10543711 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | American Society for Clinical Investigation |
record_format | MEDLINE/PubMed |
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 |
work_keys_str_mv | AT rothcarterquinnr transcriptionalprofilingofrareacantholyticdisorderssuggestscommonmechanismsofpathogenesis AT burkshopee transcriptionalprofilingofrareacantholyticdisorderssuggestscommonmechanismsofpathogenesis AT renziyou transcriptionalprofilingofrareacantholyticdisorderssuggestscommonmechanismsofpathogenesis AT koetsierjenniferl transcriptionalprofilingofrareacantholyticdisorderssuggestscommonmechanismsofpathogenesis AT tsoilamc transcriptionalprofilingofrareacantholyticdisorderssuggestscommonmechanismsofpathogenesis AT harmspaulw transcriptionalprofilingofrareacantholyticdisorderssuggestscommonmechanismsofpathogenesis AT xingxianying transcriptionalprofilingofrareacantholyticdisorderssuggestscommonmechanismsofpathogenesis AT kirmajoseph transcriptionalprofilingofrareacantholyticdisorderssuggestscommonmechanismsofpathogenesis AT harmonrobertm transcriptionalprofilingofrareacantholyticdisorderssuggestscommonmechanismsofpathogenesis AT godsellisam transcriptionalprofilingofrareacantholyticdisorderssuggestscommonmechanismsofpathogenesis AT perlabbeyl transcriptionalprofilingofrareacantholyticdisorderssuggestscommonmechanismsofpathogenesis AT gudjonssonjohanne transcriptionalprofilingofrareacantholyticdisorderssuggestscommonmechanismsofpathogenesis AT greenkathleenj transcriptionalprofilingofrareacantholyticdisorderssuggestscommonmechanismsofpathogenesis |