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A rare case of Sweet syndrome secondary to melioidosis

BACKGROUND: Melioidosis is an emerging infection in South Asia caused by Burkholderia pseudomallei with various clinical presentations that include pneumonia, bacteraemia, arthritis, and deep-seated abscesses. Various cutaneous manifestations have been described in association with melioidosis. Howe...

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Autores principales: Vithoosan, Sahathevan, Thanushah, Balendran, Piranavan, Paramarajan, Gamlaksha, Dayal, Karunatilake, Harindra, Jayanaga, Ananda
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2019
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Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6886164/
https://www.ncbi.nlm.nih.gov/pubmed/31787085
http://dx.doi.org/10.1186/s12895-019-0096-2
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author Vithoosan, Sahathevan
Thanushah, Balendran
Piranavan, Paramarajan
Gamlaksha, Dayal
Karunatilake, Harindra
Jayanaga, Ananda
author_facet Vithoosan, Sahathevan
Thanushah, Balendran
Piranavan, Paramarajan
Gamlaksha, Dayal
Karunatilake, Harindra
Jayanaga, Ananda
author_sort Vithoosan, Sahathevan
collection PubMed
description BACKGROUND: Melioidosis is an emerging infection in South Asia caused by Burkholderia pseudomallei with various clinical presentations that include pneumonia, bacteraemia, arthritis, and deep-seated abscesses. Various cutaneous manifestations have been described in association with melioidosis. However Sweet Syndrome secondary to melioidosis has not been reported in the literature. Herein we describe the first case of Sweet syndrome secondary to melioidosis. CASE PRESENTATION: A 53-year-old previously healthy Sri Lankan female presented with high-grade fever, painful oral ulcers, odynophagia and multiple bilateral cervical lymphadenopathies for 1 month. She also had a loss of appetite and weight. She had oral ulcers and bilateral blepharitis. Dermatological examination revealed multiple tender papules with a mamillated appearance and targetoid lesions with a yellowish centre over the face, upper trunk and upper limbs. She also had multiple tender subcutaneous nodules over the extensor aspect of upper limbs. Her inflammatory markers were significantly elevated. Aspirate from a submental lymph node abscess revealed the growth of Burkholderia pseudomallei. Melioidosis antibody titer was > 10,240. The histology of the skin lesions of the face and left forearm showed a prominent neutrophilic infiltrate in the dermis and the morphological features were in favour of Sweet syndrome with panniculitis. She was started on intravenous meropenem 2 g daily and showed rapid clinical improvement with the disappearance of skin lesions as well as a reduction in inflammatory markers. CONCLUSION: Sweet syndrome is an uncommon inflammatory disorder known to be associated with upper respiratory tract and gastrointestinal infections, malignancies and the use of certain drugs. Melioidosis is an emerging infection with various cutaneous manifestations. This is the first case of melioidosis causing the secondary sweet syndrome. It emphasizes the importance of considering melioidosis as a potential aetiology in patients with Sweet syndrome.
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spelling pubmed-68861642019-12-11 A rare case of Sweet syndrome secondary to melioidosis Vithoosan, Sahathevan Thanushah, Balendran Piranavan, Paramarajan Gamlaksha, Dayal Karunatilake, Harindra Jayanaga, Ananda BMC Dermatol Case Report BACKGROUND: Melioidosis is an emerging infection in South Asia caused by Burkholderia pseudomallei with various clinical presentations that include pneumonia, bacteraemia, arthritis, and deep-seated abscesses. Various cutaneous manifestations have been described in association with melioidosis. However Sweet Syndrome secondary to melioidosis has not been reported in the literature. Herein we describe the first case of Sweet syndrome secondary to melioidosis. CASE PRESENTATION: A 53-year-old previously healthy Sri Lankan female presented with high-grade fever, painful oral ulcers, odynophagia and multiple bilateral cervical lymphadenopathies for 1 month. She also had a loss of appetite and weight. She had oral ulcers and bilateral blepharitis. Dermatological examination revealed multiple tender papules with a mamillated appearance and targetoid lesions with a yellowish centre over the face, upper trunk and upper limbs. She also had multiple tender subcutaneous nodules over the extensor aspect of upper limbs. Her inflammatory markers were significantly elevated. Aspirate from a submental lymph node abscess revealed the growth of Burkholderia pseudomallei. Melioidosis antibody titer was > 10,240. The histology of the skin lesions of the face and left forearm showed a prominent neutrophilic infiltrate in the dermis and the morphological features were in favour of Sweet syndrome with panniculitis. She was started on intravenous meropenem 2 g daily and showed rapid clinical improvement with the disappearance of skin lesions as well as a reduction in inflammatory markers. CONCLUSION: Sweet syndrome is an uncommon inflammatory disorder known to be associated with upper respiratory tract and gastrointestinal infections, malignancies and the use of certain drugs. Melioidosis is an emerging infection with various cutaneous manifestations. This is the first case of melioidosis causing the secondary sweet syndrome. It emphasizes the importance of considering melioidosis as a potential aetiology in patients with Sweet syndrome. BioMed Central 2019-12-02 /pmc/articles/PMC6886164/ /pubmed/31787085 http://dx.doi.org/10.1186/s12895-019-0096-2 Text en © The Author(s). 2019 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
Vithoosan, Sahathevan
Thanushah, Balendran
Piranavan, Paramarajan
Gamlaksha, Dayal
Karunatilake, Harindra
Jayanaga, Ananda
A rare case of Sweet syndrome secondary to melioidosis
title A rare case of Sweet syndrome secondary to melioidosis
title_full A rare case of Sweet syndrome secondary to melioidosis
title_fullStr A rare case of Sweet syndrome secondary to melioidosis
title_full_unstemmed A rare case of Sweet syndrome secondary to melioidosis
title_short A rare case of Sweet syndrome secondary to melioidosis
title_sort rare case of sweet syndrome secondary to melioidosis
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6886164/
https://www.ncbi.nlm.nih.gov/pubmed/31787085
http://dx.doi.org/10.1186/s12895-019-0096-2
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