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A Case of Spondyloarthritis in Patient Affected by Unicentric Castleman's Disease Effectively Managed with Surgery Resection and Tocilizumab Treatment

A 38-year-old woman was referred to our hospital for rheumatologic manifestations (migrant arthritis and tenosynovitis), without psoriasis or family history of psoriasis, gastroenteric manifestations, or recent genitourinary infections. The instrumental and laboratory tests have suggested a diagnosi...

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Autores principales: Filippini, M., Cartella, S., Bonzanini, O., Morello, E., Tincani, A.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5828050/
https://www.ncbi.nlm.nih.gov/pubmed/29610697
http://dx.doi.org/10.1155/2018/5067239
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author Filippini, M.
Cartella, S.
Bonzanini, O.
Morello, E.
Tincani, A.
author_facet Filippini, M.
Cartella, S.
Bonzanini, O.
Morello, E.
Tincani, A.
author_sort Filippini, M.
collection PubMed
description A 38-year-old woman was referred to our hospital for rheumatologic manifestations (migrant arthritis and tenosynovitis), without psoriasis or family history of psoriasis, gastroenteric manifestations, or recent genitourinary infections. The instrumental and laboratory tests have suggested a diagnosis of undifferentiated seronegative HLA-B27-positive spondyloarthritis with predominantly peripheral involvement. The symptoms were very severe and resistant to anti-inflammatory drugs and steroids. She had a history of hyaline-vascular unicentric Castleman's disease (HBV, HIV, and HHV-8 negative) treated with surgery resection. After a first pharmacological attempt with sulfasalazine (suspended for urticarial rash), we managed the patient with monotherapy tocilizumab 8 mg/kg, with full response of rheumatologic manifestations. The efficacy of tocilizumab was confirmed even after a follow-up of three years. Our experience seems to describe a new late-onset autoimmune disease (only 21 cases described in literature) potentially related to Castleman's disease. The patient experienced marked improvement from IL-6-based therapy (tocilizumab).
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spelling pubmed-58280502018-04-02 A Case of Spondyloarthritis in Patient Affected by Unicentric Castleman's Disease Effectively Managed with Surgery Resection and Tocilizumab Treatment Filippini, M. Cartella, S. Bonzanini, O. Morello, E. Tincani, A. Case Rep Rheumatol Case Report A 38-year-old woman was referred to our hospital for rheumatologic manifestations (migrant arthritis and tenosynovitis), without psoriasis or family history of psoriasis, gastroenteric manifestations, or recent genitourinary infections. The instrumental and laboratory tests have suggested a diagnosis of undifferentiated seronegative HLA-B27-positive spondyloarthritis with predominantly peripheral involvement. The symptoms were very severe and resistant to anti-inflammatory drugs and steroids. She had a history of hyaline-vascular unicentric Castleman's disease (HBV, HIV, and HHV-8 negative) treated with surgery resection. After a first pharmacological attempt with sulfasalazine (suspended for urticarial rash), we managed the patient with monotherapy tocilizumab 8 mg/kg, with full response of rheumatologic manifestations. The efficacy of tocilizumab was confirmed even after a follow-up of three years. Our experience seems to describe a new late-onset autoimmune disease (only 21 cases described in literature) potentially related to Castleman's disease. The patient experienced marked improvement from IL-6-based therapy (tocilizumab). Hindawi 2018-01-23 /pmc/articles/PMC5828050/ /pubmed/29610697 http://dx.doi.org/10.1155/2018/5067239 Text en Copyright © 2018 M. Filippini et al. http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Filippini, M.
Cartella, S.
Bonzanini, O.
Morello, E.
Tincani, A.
A Case of Spondyloarthritis in Patient Affected by Unicentric Castleman's Disease Effectively Managed with Surgery Resection and Tocilizumab Treatment
title A Case of Spondyloarthritis in Patient Affected by Unicentric Castleman's Disease Effectively Managed with Surgery Resection and Tocilizumab Treatment
title_full A Case of Spondyloarthritis in Patient Affected by Unicentric Castleman's Disease Effectively Managed with Surgery Resection and Tocilizumab Treatment
title_fullStr A Case of Spondyloarthritis in Patient Affected by Unicentric Castleman's Disease Effectively Managed with Surgery Resection and Tocilizumab Treatment
title_full_unstemmed A Case of Spondyloarthritis in Patient Affected by Unicentric Castleman's Disease Effectively Managed with Surgery Resection and Tocilizumab Treatment
title_short A Case of Spondyloarthritis in Patient Affected by Unicentric Castleman's Disease Effectively Managed with Surgery Resection and Tocilizumab Treatment
title_sort case of spondyloarthritis in patient affected by unicentric castleman's disease effectively managed with surgery resection and tocilizumab treatment
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5828050/
https://www.ncbi.nlm.nih.gov/pubmed/29610697
http://dx.doi.org/10.1155/2018/5067239
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