Seronegative Arthritis and Whipple Disease: Risk of Misdiagnosis in the Era of Biologic Agents

We report 2 cases of Whipple disease (WD), previously diagnosed as seronegative polyarthritis and treated for several years with immunosuppressive agents, accordingly. Both cases had been treated over years with cDMARDs and bDMARDs. The first patient was a 48-year-old male, who developed a life-thre...

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Main Authors: Luca Quartuccio, Ivan Giovannini, Stefano Pizzolitto, Maurizio Scarpa, Salvatore De Vita
Format: Article
Language:English
Published: Wiley 2019-01-01
Series:Case Reports in Rheumatology
Online Access:http://dx.doi.org/10.1155/2019/3410468
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author Luca Quartuccio
Ivan Giovannini
Stefano Pizzolitto
Maurizio Scarpa
Salvatore De Vita
author_facet Luca Quartuccio
Ivan Giovannini
Stefano Pizzolitto
Maurizio Scarpa
Salvatore De Vita
author_sort Luca Quartuccio
collection DOAJ
description We report 2 cases of Whipple disease (WD), previously diagnosed as seronegative polyarthritis and treated for several years with immunosuppressive agents, accordingly. Both cases had been treated over years with cDMARDs and bDMARDs. The first patient was a 48-year-old male, who developed a life-threatening disease characterized by fever, significant weight loss, and bloody diarrhoea, supported with RBC transfusions. The second patient was a 55-year-old man, presenting with arthritis, fever, serositis, lymphadenopathy, thoracic rash, and systemic inflammation; at the beginning he was diagnosed as adult onset Still’s disease. He was treated with steroids and antitumour necrosis factor agents, but showed no improvement. Both patients were eventually treated with antimicrobial therapy for WD with dramatic improvement and no clinical relapse in 6 months. This paper reviews the literature on WD mimicking chronic inflammatory arthritis. WD may lead to chronic seronegative arthritis that might often be misrecognized. Importantly, patients treated with bDMARDs and glucocorticoids might develop a life-threatening disease. Therefore, WD should be suspected and excluded in patients showing resistance or frequent recurrence of chronic arthritis, if seronegative, under treatment with bDMARDs, especially in the presence of new, unexpected sign and/or symptoms.
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language English
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spelling doaj-art-4fcb92c587d94c7da8f5b05ab7ae5d012025-02-03T06:11:25ZengWileyCase Reports in Rheumatology2090-68892090-68972019-01-01201910.1155/2019/34104683410468Seronegative Arthritis and Whipple Disease: Risk of Misdiagnosis in the Era of Biologic AgentsLuca Quartuccio0Ivan Giovannini1Stefano Pizzolitto2Maurizio Scarpa3Salvatore De Vita4Rheumatology Clinic, Department of Medical Area, Academic Hospital Santa Maria della Misericordia, University of Udine, Udine, ItalyRheumatology Clinic, Department of Medical Area, Academic Hospital Santa Maria della Misericordia, University of Udine, Udine, ItalyInstitute of Pathology, Academic Hospital Santa Maria della Misericordia, Udine, ItalyRare Diseases Regional Coordinating Centre, Academic Hospital Santa Maria della Misericordia, Udine, ItalyRheumatology Clinic, Department of Medical Area, Academic Hospital Santa Maria della Misericordia, University of Udine, Udine, ItalyWe report 2 cases of Whipple disease (WD), previously diagnosed as seronegative polyarthritis and treated for several years with immunosuppressive agents, accordingly. Both cases had been treated over years with cDMARDs and bDMARDs. The first patient was a 48-year-old male, who developed a life-threatening disease characterized by fever, significant weight loss, and bloody diarrhoea, supported with RBC transfusions. The second patient was a 55-year-old man, presenting with arthritis, fever, serositis, lymphadenopathy, thoracic rash, and systemic inflammation; at the beginning he was diagnosed as adult onset Still’s disease. He was treated with steroids and antitumour necrosis factor agents, but showed no improvement. Both patients were eventually treated with antimicrobial therapy for WD with dramatic improvement and no clinical relapse in 6 months. This paper reviews the literature on WD mimicking chronic inflammatory arthritis. WD may lead to chronic seronegative arthritis that might often be misrecognized. Importantly, patients treated with bDMARDs and glucocorticoids might develop a life-threatening disease. Therefore, WD should be suspected and excluded in patients showing resistance or frequent recurrence of chronic arthritis, if seronegative, under treatment with bDMARDs, especially in the presence of new, unexpected sign and/or symptoms.http://dx.doi.org/10.1155/2019/3410468
spellingShingle Luca Quartuccio
Ivan Giovannini
Stefano Pizzolitto
Maurizio Scarpa
Salvatore De Vita
Seronegative Arthritis and Whipple Disease: Risk of Misdiagnosis in the Era of Biologic Agents
Case Reports in Rheumatology
title Seronegative Arthritis and Whipple Disease: Risk of Misdiagnosis in the Era of Biologic Agents
title_full Seronegative Arthritis and Whipple Disease: Risk of Misdiagnosis in the Era of Biologic Agents
title_fullStr Seronegative Arthritis and Whipple Disease: Risk of Misdiagnosis in the Era of Biologic Agents
title_full_unstemmed Seronegative Arthritis and Whipple Disease: Risk of Misdiagnosis in the Era of Biologic Agents
title_short Seronegative Arthritis and Whipple Disease: Risk of Misdiagnosis in the Era of Biologic Agents
title_sort seronegative arthritis and whipple disease risk of misdiagnosis in the era of biologic agents
url http://dx.doi.org/10.1155/2019/3410468
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