Expression of polydom in dermal neurofibroma and surrounding dermis in von Recklinghausen's disease.


Journal

Journal of dermatological science
ISSN: 1873-569X
Titre abrégé: J Dermatol Sci
Pays: Netherlands
ID NLM: 9011485

Informations de publication

Date de publication:
Nov 2019
Historique:
received: 03 12 2018
revised: 06 08 2019
accepted: 12 09 2019
pubmed: 2 10 2019
medline: 23 6 2020
entrez: 2 10 2019
Statut: ppublish

Résumé

Neurofibromas in von Recklinghausen's disease (vRD) can develop in the dermis. Therefore, we hypothesized that a dermal niche exists that promotes the development of these neurofibromas in subjects with vRD. The purpose of this study is to examine the function of polydom, known as a ligand for integrin, mediating cell adhesion, and expressed in mouse nerve tissue, in promotion of neurofibroma. Molecular, transcriptome and immunohistochemical analysis were performed to investigate the association between polydom expression and neurofibroma development. Polydom mRNA levels were significantly higher in neurofibroma tissue than in control tissue. Quantitative reverse transcription-polymerase chain reaction (qRT-PCR) analysis of RNA purified from primary cultured dermal neurofibroma cells demonstrated significantly higher polydom mRNA expression in cells derived from the surrounding dermis of neurofibromas compared to those from normal human dermal fibroblasts. RNA sequencing was used to compare gene expression between cultured cells derived from dermal neurofibroma-surrounding tissue with or without polydom knockdown. Subsequent gene ontology assays revealed that expression of integrinβ8 (ITGB8), a factor that releases transforming growth factor-β (TGF-β) from pro-TGF-β, was downregulated following polydom knockdown, suggesting upregulation of polydom-mediated TGF-β production. Furthermore, we observed a strong association between polydom expression and the increase in platelet-derived growth factor B (PDGFB) expression in primary cultured cells from the surrounding dermis of neurofibromas exposed to TGF-β1. Our results suggest that increased polydom expression in the dermis surrounding neurofibromas may promote dermal neurofibroma development by activating the TGF-β signaling pathway.

Sections du résumé

BACKGROUND BACKGROUND
Neurofibromas in von Recklinghausen's disease (vRD) can develop in the dermis. Therefore, we hypothesized that a dermal niche exists that promotes the development of these neurofibromas in subjects with vRD.
OBJECTIVE OBJECTIVE
The purpose of this study is to examine the function of polydom, known as a ligand for integrin, mediating cell adhesion, and expressed in mouse nerve tissue, in promotion of neurofibroma.
METHODS METHODS
Molecular, transcriptome and immunohistochemical analysis were performed to investigate the association between polydom expression and neurofibroma development.
RESULTS RESULTS
Polydom mRNA levels were significantly higher in neurofibroma tissue than in control tissue. Quantitative reverse transcription-polymerase chain reaction (qRT-PCR) analysis of RNA purified from primary cultured dermal neurofibroma cells demonstrated significantly higher polydom mRNA expression in cells derived from the surrounding dermis of neurofibromas compared to those from normal human dermal fibroblasts. RNA sequencing was used to compare gene expression between cultured cells derived from dermal neurofibroma-surrounding tissue with or without polydom knockdown. Subsequent gene ontology assays revealed that expression of integrinβ8 (ITGB8), a factor that releases transforming growth factor-β (TGF-β) from pro-TGF-β, was downregulated following polydom knockdown, suggesting upregulation of polydom-mediated TGF-β production. Furthermore, we observed a strong association between polydom expression and the increase in platelet-derived growth factor B (PDGFB) expression in primary cultured cells from the surrounding dermis of neurofibromas exposed to TGF-β1.
CONCLUSION CONCLUSIONS
Our results suggest that increased polydom expression in the dermis surrounding neurofibromas may promote dermal neurofibroma development by activating the TGF-β signaling pathway.

Identifiants

pubmed: 31570272
pii: S0923-1811(19)30295-6
doi: 10.1016/j.jdermsci.2019.09.005
pii:
doi:

Substances chimiques

Cell Adhesion Molecules 0
Integrin beta Chains 0
Proto-Oncogene Proteins c-sis 0
RNA, Small Interfering 0
SVEP1 protein, human 0
TGFB1 protein, human 0
Transforming Growth Factor beta 0
Transforming Growth Factor beta1 0
integrin beta8 0

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

73-80

Informations de copyright

Copyright © 2019. Published by Elsevier B.V.

Auteurs

Tomo Kamitani (T)

Dermatology, Department of Integrated Medicine, Graduate School of Medicine, Osaka University, Osaka, Japan.

Hiroyuki Murota (H)

Dermatology, Department of Integrated Medicine, Graduate School of Medicine, Osaka University, Osaka, Japan; Department of Dermatology, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki, Japan. Electronic address: h-murota@nagasaki-u.ac.jp.

Noriko Arase (N)

Dermatology, Department of Integrated Medicine, Graduate School of Medicine, Osaka University, Osaka, Japan.

Mari Wataya-Kaneda (M)

Dermatology, Department of Integrated Medicine, Graduate School of Medicine, Osaka University, Osaka, Japan.

Ryoko Sato-Nishiuchi (R)

Division of Matrixome Research and Application, Institute for Protein Research, Osaka University, Osaka, Japan.

Kiyotoshi Sekiguchi (K)

Division of Matrixome Research and Application, Institute for Protein Research, Osaka University, Osaka, Japan.

Daisuke Okuzaki (D)

Genome Information Research Center, Research Institute for Microbial Diseases, Osaka University, Osaka, Japan.

Daisuke Motooka (D)

Genome Information Research Center, Research Institute for Microbial Diseases, Osaka University, Osaka, Japan.

Ichiro Katayama (I)

Dermatology, Department of Integrated Medicine, Graduate School of Medicine, Osaka University, Osaka, Japan; Department of Pigmentation Research and Therapeutics Graduate School of Medicine, Osaka City University, Osaka, Japan.

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Classifications MeSH