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Sweet’s Syndrome Following Therapy with Hydroxychloroquine in a Patient Affected with Elderly-Onset Primary Sjogren’s Syndrome

Sweet’s syndrome is an uncommon skin disease characterized by painful polymorphic lesions associated with fever and neutrophilia. When biopsied, these lesions reveal a diffuse infiltrate of mature neutrophils in the papillary dermis. Several drugs can induce Sweet’s syndrome (so-called drug-induced...

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Autores principales: Manzo, Ciro, Pollio, Nazareno, Natale, Maria
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6963767/
https://www.ncbi.nlm.nih.gov/pubmed/31731586
http://dx.doi.org/10.3390/medicines6040111
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author Manzo, Ciro
Pollio, Nazareno
Natale, Maria
author_facet Manzo, Ciro
Pollio, Nazareno
Natale, Maria
author_sort Manzo, Ciro
collection PubMed
description Sweet’s syndrome is an uncommon skin disease characterized by painful polymorphic lesions associated with fever and neutrophilia. When biopsied, these lesions reveal a diffuse infiltrate of mature neutrophils in the papillary dermis. Several drugs can induce Sweet’s syndrome (so-called drug-induced Sweet’s syndrome (DISS)) but reports of DISS associated with hydroxychloroquine (HCQ) are exceptionally limited. A 72-year-old Caucasian female patient with elderly-onset primary Sjogren’s syndrome (EOpSS) but low disease activity presented with an abrupt onset of painful nodular and papular erythematous skin lesions after two weeks of therapy with HCQ 400 mg. A histological examination revealed a diffuse infiltrate of mature neutrophils in the papillary dermis, without vasculitis. After therapy with 25 mg/day prednisone and HCQ withdrawal, the cutaneous manifestations disappeared. When prednisone was permanently discontinued, the primary Sjogren’s syndrome (pSS) manifestations worsened and therapy with HCQ 200 mg was reintroduced. In a few days, the same skin lesions reappeared. Withdrawal of HCQ and a new cycle of prednisone resulted in their permanent disappearance. We reported a case of DISS following therapy with HCQ in a female patient affected by EOpSS. According to a literature review, this is the first report of this association.
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spelling pubmed-69637672020-01-27 Sweet’s Syndrome Following Therapy with Hydroxychloroquine in a Patient Affected with Elderly-Onset Primary Sjogren’s Syndrome Manzo, Ciro Pollio, Nazareno Natale, Maria Medicines (Basel) Case Report Sweet’s syndrome is an uncommon skin disease characterized by painful polymorphic lesions associated with fever and neutrophilia. When biopsied, these lesions reveal a diffuse infiltrate of mature neutrophils in the papillary dermis. Several drugs can induce Sweet’s syndrome (so-called drug-induced Sweet’s syndrome (DISS)) but reports of DISS associated with hydroxychloroquine (HCQ) are exceptionally limited. A 72-year-old Caucasian female patient with elderly-onset primary Sjogren’s syndrome (EOpSS) but low disease activity presented with an abrupt onset of painful nodular and papular erythematous skin lesions after two weeks of therapy with HCQ 400 mg. A histological examination revealed a diffuse infiltrate of mature neutrophils in the papillary dermis, without vasculitis. After therapy with 25 mg/day prednisone and HCQ withdrawal, the cutaneous manifestations disappeared. When prednisone was permanently discontinued, the primary Sjogren’s syndrome (pSS) manifestations worsened and therapy with HCQ 200 mg was reintroduced. In a few days, the same skin lesions reappeared. Withdrawal of HCQ and a new cycle of prednisone resulted in their permanent disappearance. We reported a case of DISS following therapy with HCQ in a female patient affected by EOpSS. According to a literature review, this is the first report of this association. MDPI 2019-11-15 /pmc/articles/PMC6963767/ /pubmed/31731586 http://dx.doi.org/10.3390/medicines6040111 Text en © 2019 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (http://creativecommons.org/licenses/by/4.0/).
spellingShingle Case Report
Manzo, Ciro
Pollio, Nazareno
Natale, Maria
Sweet’s Syndrome Following Therapy with Hydroxychloroquine in a Patient Affected with Elderly-Onset Primary Sjogren’s Syndrome
title Sweet’s Syndrome Following Therapy with Hydroxychloroquine in a Patient Affected with Elderly-Onset Primary Sjogren’s Syndrome
title_full Sweet’s Syndrome Following Therapy with Hydroxychloroquine in a Patient Affected with Elderly-Onset Primary Sjogren’s Syndrome
title_fullStr Sweet’s Syndrome Following Therapy with Hydroxychloroquine in a Patient Affected with Elderly-Onset Primary Sjogren’s Syndrome
title_full_unstemmed Sweet’s Syndrome Following Therapy with Hydroxychloroquine in a Patient Affected with Elderly-Onset Primary Sjogren’s Syndrome
title_short Sweet’s Syndrome Following Therapy with Hydroxychloroquine in a Patient Affected with Elderly-Onset Primary Sjogren’s Syndrome
title_sort sweet’s syndrome following therapy with hydroxychloroquine in a patient affected with elderly-onset primary sjogren’s syndrome
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6963767/
https://www.ncbi.nlm.nih.gov/pubmed/31731586
http://dx.doi.org/10.3390/medicines6040111
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