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Efficacy of gentamicin 0.3% solution of oral erosions healing in patients with severe generalized recessive dystrophic epidermolysis bullosa and its impact on the expression of type VII collagen

INTRODUCTION: Epidermolysis bullosa (EB) is a rare genetic skin disorder inherited either in autosomal recessive (AR) or autosomal dominant (AD) manner and characterized by blistering of the skin and mucous membranes. According to a subtype of EB, the oral manifestations and dental involvement vary...

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Autores principales: Osipowicz, Katarzyna, Wychowanski, Piotr, Nieckula, Pawel, Shamsa, Sara, Wertheim-Tysarowska, Katarzyna, Wozniak, Katarzyna, Kowalewski, Cezary
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Termedia Publishing House 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8802963/
https://www.ncbi.nlm.nih.gov/pubmed/35126004
http://dx.doi.org/10.5114/ada.2020.97072
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author Osipowicz, Katarzyna
Wychowanski, Piotr
Nieckula, Pawel
Shamsa, Sara
Wertheim-Tysarowska, Katarzyna
Wozniak, Katarzyna
Kowalewski, Cezary
author_facet Osipowicz, Katarzyna
Wychowanski, Piotr
Nieckula, Pawel
Shamsa, Sara
Wertheim-Tysarowska, Katarzyna
Wozniak, Katarzyna
Kowalewski, Cezary
author_sort Osipowicz, Katarzyna
collection PubMed
description INTRODUCTION: Epidermolysis bullosa (EB) is a rare genetic skin disorder inherited either in autosomal recessive (AR) or autosomal dominant (AD) manner and characterized by blistering of the skin and mucous membranes. According to a subtype of EB, the oral manifestations and dental involvement vary in frequency and in severity. The most severe dental problems occur in patients with junctional epidermolysis bullosa (JEB) and severe generalized dystrophic epidermolysis bullosa (RDEB) and involve enamel erosion and development of blisters followed by painful oral wounds. Oral mucosa lesions decrease patients’ quality of life and may contribute to difficulties in nutrition leading to cachexia. AIM: Assessment of efficacy of gentamicin 0.3% solution in the healing and preventing of oral erosions in patients with RDEB and evaluating its impact on the expression of type VII collagen. MATERIAL AND METHODS: The study included four female patients with RDEB, aged 16–42 who show different mutations in the COL7A1 gene and were administered the mouth rinse two times daily with a solution of 0.3% gentamycin for 4 consecutive weeks. Prior to and at the end of the study, the samples from oral mucosa were collected to estimate the expression of type VII collagen by immunofluorescence test. RESULTS: The clinical improvement of oral wounds healing and reduced number of new blisters and mucous membrane soreness as well as partial re-expression of type VII collagen was observed in all studied patients. CONCLUSIONS: Topical gentamicin therapy of oral cavity in RDEB patients resulted in clinical improvement of mucosal lesions and re-expression of collagen type VII.
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spelling pubmed-88029632022-02-04 Efficacy of gentamicin 0.3% solution of oral erosions healing in patients with severe generalized recessive dystrophic epidermolysis bullosa and its impact on the expression of type VII collagen Osipowicz, Katarzyna Wychowanski, Piotr Nieckula, Pawel Shamsa, Sara Wertheim-Tysarowska, Katarzyna Wozniak, Katarzyna Kowalewski, Cezary Postepy Dermatol Alergol Original Paper INTRODUCTION: Epidermolysis bullosa (EB) is a rare genetic skin disorder inherited either in autosomal recessive (AR) or autosomal dominant (AD) manner and characterized by blistering of the skin and mucous membranes. According to a subtype of EB, the oral manifestations and dental involvement vary in frequency and in severity. The most severe dental problems occur in patients with junctional epidermolysis bullosa (JEB) and severe generalized dystrophic epidermolysis bullosa (RDEB) and involve enamel erosion and development of blisters followed by painful oral wounds. Oral mucosa lesions decrease patients’ quality of life and may contribute to difficulties in nutrition leading to cachexia. AIM: Assessment of efficacy of gentamicin 0.3% solution in the healing and preventing of oral erosions in patients with RDEB and evaluating its impact on the expression of type VII collagen. MATERIAL AND METHODS: The study included four female patients with RDEB, aged 16–42 who show different mutations in the COL7A1 gene and were administered the mouth rinse two times daily with a solution of 0.3% gentamycin for 4 consecutive weeks. Prior to and at the end of the study, the samples from oral mucosa were collected to estimate the expression of type VII collagen by immunofluorescence test. RESULTS: The clinical improvement of oral wounds healing and reduced number of new blisters and mucous membrane soreness as well as partial re-expression of type VII collagen was observed in all studied patients. CONCLUSIONS: Topical gentamicin therapy of oral cavity in RDEB patients resulted in clinical improvement of mucosal lesions and re-expression of collagen type VII. Termedia Publishing House 2020-07-14 2021-12 /pmc/articles/PMC8802963/ /pubmed/35126004 http://dx.doi.org/10.5114/ada.2020.97072 Text en Copyright: © 2021 Termedia Sp. z o. o. https://creativecommons.org/licenses/by-nc-sa/4.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International (CC BY-NC-SA 4.0) License, allowing third parties to copy and redistribute the material in any medium or format and to remix, transform, and build upon the material, provided the original work is properly cited and states its license.
spellingShingle Original Paper
Osipowicz, Katarzyna
Wychowanski, Piotr
Nieckula, Pawel
Shamsa, Sara
Wertheim-Tysarowska, Katarzyna
Wozniak, Katarzyna
Kowalewski, Cezary
Efficacy of gentamicin 0.3% solution of oral erosions healing in patients with severe generalized recessive dystrophic epidermolysis bullosa and its impact on the expression of type VII collagen
title Efficacy of gentamicin 0.3% solution of oral erosions healing in patients with severe generalized recessive dystrophic epidermolysis bullosa and its impact on the expression of type VII collagen
title_full Efficacy of gentamicin 0.3% solution of oral erosions healing in patients with severe generalized recessive dystrophic epidermolysis bullosa and its impact on the expression of type VII collagen
title_fullStr Efficacy of gentamicin 0.3% solution of oral erosions healing in patients with severe generalized recessive dystrophic epidermolysis bullosa and its impact on the expression of type VII collagen
title_full_unstemmed Efficacy of gentamicin 0.3% solution of oral erosions healing in patients with severe generalized recessive dystrophic epidermolysis bullosa and its impact on the expression of type VII collagen
title_short Efficacy of gentamicin 0.3% solution of oral erosions healing in patients with severe generalized recessive dystrophic epidermolysis bullosa and its impact on the expression of type VII collagen
title_sort efficacy of gentamicin 0.3% solution of oral erosions healing in patients with severe generalized recessive dystrophic epidermolysis bullosa and its impact on the expression of type vii collagen
topic Original Paper
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8802963/
https://www.ncbi.nlm.nih.gov/pubmed/35126004
http://dx.doi.org/10.5114/ada.2020.97072
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