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A case report of erythroderma in a patient with borderline leprosy on reversal reaction: a result of the exacerbated reaction?
BACKGROUND: Erythroderma is characterized by erythema and scaling affecting more than 90% of the body surface area. Inflammatory, neoplastic and, more rarely, infectious diseases may culminate with erythroderma. Diagnosis of the underlying disorder is therefore crucial to institute the appropriate t...
Autores principales: | , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
BioMed Central
2017
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5738805/ https://www.ncbi.nlm.nih.gov/pubmed/29262820 http://dx.doi.org/10.1186/s12895-017-0068-3 |
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author | Miyashiro, Denis Vieira, Ana Paula Trindade, Maria Angela Bianconcini Avancini, João Sanches, José Antonio Benard, Gil |
author_facet | Miyashiro, Denis Vieira, Ana Paula Trindade, Maria Angela Bianconcini Avancini, João Sanches, José Antonio Benard, Gil |
author_sort | Miyashiro, Denis |
collection | PubMed |
description | BACKGROUND: Erythroderma is characterized by erythema and scaling affecting more than 90% of the body surface area. Inflammatory, neoplastic and, more rarely, infectious diseases may culminate with erythroderma. Diagnosis of the underlying disorder is therefore crucial to institute the appropriate therapy. Leprosy is a chronic infectious disease that is endemic in Brazil. Here we present an unusual case of leprosy and reversal reaction causing erythroderma, and we discuss the underlying immunological mechanisms which could contribute to the generalized skin inflammation. CASE PRESENTATION: We report a case of a patient with reversal reaction (RR) in borderline borderline leprosy presenting with erythroderma and neural disabilities. Histopathology of the skin showed regular acanthosis and spongiosis in the epidermis and, in the dermis, compact epithelioid granulomas as well as grouped and isolated bacilli. This duality probably reflects the transition from an anergic/multibacillary state to a state of more effective immunity and bacillary control, typical of RR. Leprosy was successfully treated with WHO’s multidrug therapy, plus prednisone for controlling the RR; the erythroderma resolved in parallel with this treatment. Immunologic studies showed in situ predominance of IFNγ + over IL-4+ lymphocytes and of IL-17+ over Foxp3+ lymphocytes, suggesting an exacerbated Th-1/Th-17 immunoreactivity and poor Th-2 and regulatory T-cell responses. Circulating Tregs were also diminished. We hypothesize that the flare-up of anti-mycobacteria immunoreactivity that underlies RR may have triggered the intense inflammatory skin lesions that culminated with erythroderma. CONCLUSIONS: This case report highlights the importance of thorough clinical examination of erythrodermic patients in search for its etiology and suggests that an intense and probably uncontrolled leprosy RR can culminate in the development of erythroderma. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (10.1186/s12895-017-0068-3) contains supplementary material, which is available to authorized users. |
format | Online Article Text |
id | pubmed-5738805 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2017 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-57388052018-01-02 A case report of erythroderma in a patient with borderline leprosy on reversal reaction: a result of the exacerbated reaction? Miyashiro, Denis Vieira, Ana Paula Trindade, Maria Angela Bianconcini Avancini, João Sanches, José Antonio Benard, Gil BMC Dermatol Case Report BACKGROUND: Erythroderma is characterized by erythema and scaling affecting more than 90% of the body surface area. Inflammatory, neoplastic and, more rarely, infectious diseases may culminate with erythroderma. Diagnosis of the underlying disorder is therefore crucial to institute the appropriate therapy. Leprosy is a chronic infectious disease that is endemic in Brazil. Here we present an unusual case of leprosy and reversal reaction causing erythroderma, and we discuss the underlying immunological mechanisms which could contribute to the generalized skin inflammation. CASE PRESENTATION: We report a case of a patient with reversal reaction (RR) in borderline borderline leprosy presenting with erythroderma and neural disabilities. Histopathology of the skin showed regular acanthosis and spongiosis in the epidermis and, in the dermis, compact epithelioid granulomas as well as grouped and isolated bacilli. This duality probably reflects the transition from an anergic/multibacillary state to a state of more effective immunity and bacillary control, typical of RR. Leprosy was successfully treated with WHO’s multidrug therapy, plus prednisone for controlling the RR; the erythroderma resolved in parallel with this treatment. Immunologic studies showed in situ predominance of IFNγ + over IL-4+ lymphocytes and of IL-17+ over Foxp3+ lymphocytes, suggesting an exacerbated Th-1/Th-17 immunoreactivity and poor Th-2 and regulatory T-cell responses. Circulating Tregs were also diminished. We hypothesize that the flare-up of anti-mycobacteria immunoreactivity that underlies RR may have triggered the intense inflammatory skin lesions that culminated with erythroderma. CONCLUSIONS: This case report highlights the importance of thorough clinical examination of erythrodermic patients in search for its etiology and suggests that an intense and probably uncontrolled leprosy RR can culminate in the development of erythroderma. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (10.1186/s12895-017-0068-3) contains supplementary material, which is available to authorized users. BioMed Central 2017-12-20 /pmc/articles/PMC5738805/ /pubmed/29262820 http://dx.doi.org/10.1186/s12895-017-0068-3 Text en © The Author(s). 2017 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. |
spellingShingle | Case Report Miyashiro, Denis Vieira, Ana Paula Trindade, Maria Angela Bianconcini Avancini, João Sanches, José Antonio Benard, Gil A case report of erythroderma in a patient with borderline leprosy on reversal reaction: a result of the exacerbated reaction? |
title | A case report of erythroderma in a patient with borderline leprosy on reversal reaction: a result of the exacerbated reaction? |
title_full | A case report of erythroderma in a patient with borderline leprosy on reversal reaction: a result of the exacerbated reaction? |
title_fullStr | A case report of erythroderma in a patient with borderline leprosy on reversal reaction: a result of the exacerbated reaction? |
title_full_unstemmed | A case report of erythroderma in a patient with borderline leprosy on reversal reaction: a result of the exacerbated reaction? |
title_short | A case report of erythroderma in a patient with borderline leprosy on reversal reaction: a result of the exacerbated reaction? |
title_sort | case report of erythroderma in a patient with borderline leprosy on reversal reaction: a result of the exacerbated reaction? |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5738805/ https://www.ncbi.nlm.nih.gov/pubmed/29262820 http://dx.doi.org/10.1186/s12895-017-0068-3 |
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