Fever of unknown origin with seronegative spondyloarthropathy: an atypical manifestation of Whipple's disease

Várvölgyi, Csaba; Bubán, T.; Szakáll, S; Hargitai, Zoltán; Galuska, László; Jeney, Csaba; Kakuk, G.; Gaal, José

doi:10.1136/ard.61.4.377

Cited by 14 publications

(6 citation statements)

References 13 publications

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“…However, person-to-person transmission through the oral-oral route cannot be ruled out. 3 Whipple’s disease often ends up in chronic seronegative arthritis that would be often misdiagnosed. 4 , 5 Interestingly, 1.58% of patients with unexplained arthritis and no other evidence of Whipple’s disease were positive for T. whipplei .…”

Section: Discussionmentioning

confidence: 99%

Whipple’s Disease; An Overlooked Diagnosis

Kerollos

Taha

2023

Middle East J Dig Dis

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Whipple disease is a rare multisystem inflammatory disease. Because fewer than 1000 reported cases have been described, clinical experience with this disorder is sparse. We are reporting a case of a 46-year-old man who presented with fever, weight loss, and polyarthralgia for 2 months, and 1 month of diarrhea. The patient was thoroughly investigated for collagen diseases and COVID-19, with no definite diagnosis. A therapeutic trial by immunosuppressive drugs provided partial remission followed by a marked rebound of the symptoms. His occult blood in stool was positive and subsequent upper endoscopy with proximal small intestinal biopsies showed the pathological features of Whipple’s disease. The patient showed a dramatic improvement following treatment with ceftriaxone and trimethoprim-sulfamethoxazole. Despite the rarity of Whipple’s disease, its course mimics many rheumatological diseases, inflammatory bowel disease, and COVID-19 disease. It should always be a part of the differential diagnosis of obscure polyarthralgia and chronic diarrhea.

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Section: Discussionmentioning

confidence: 99%

Whipple’s Disease; An Overlooked Diagnosis

Kerollos

Taha

2023

Middle East J Dig Dis

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“…Articular involvement often characterizes the onset of WD. The mean time from the development of articular symptoms to the diagnosis is almost 7 years [8], but there are some reports focusing the attention on the very delay of the diagnosis of WD, in the absence of gastrointestinal symptoms [14, 15]. Arthritis has been reported in 41%–61% of cases [8].…”

Section: Discussionmentioning

confidence: 99%

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

Quartuccio

Giovannini

Pizzolitto

et al. 2019

Case Reports in Rheumatology

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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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“…This unusual case highlights that Kawasaki disease should not be discounted in adult patients with features suggestive of spondylarthropathy. Indeed, conjunctival hyperemia may be confused with uveitis, Kawasaki eruptions with atypical psoriasis (which are likely [9]), distal edema with edema associated with some peripheral spondylarthropathy [10], and fever with systemic manifestations sometimes observed in patients with ankylosing spondylitis [11], reactive arthritis, or crippling infections linked to unusual germs, including Tropheryma whipplei [12].…”

Section: Discussionmentioning

confidence: 99%

Chronic, eventually fatal, Kawasaki-like disease in an adult with spondylarthropathy responding to IVIG therapy

et al. 2009

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We report on an unusual case of a 40-year-old Caucasian male displaying severe Kawasaki-like symptoms. The disease lasted for seven years before diffuse coronary aneurysms occurred, leading to the patient's death, despite ongoing treatment by intravenous immunoglobulins (IVIGs). The patient had also been suffering from a disabling inflammation of the spine, which was reported to have started at the onset of the disorder. Whereas neither NSAIDS, nor high doses corticosteroids, or anti-TNF drugs had a clear effect, the clinical features of spinal inflammation were highly sensitive to IVIGs, and were attributed definitively to HLA-B27-negative axial spondylarthropathy after bone scan and magnetic resonance imaging disclosed typical enthesitis of both heels and bilateral sacroiliitis.

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Fever of unknown origin with seronegative spondyloarthropathy: an atypical manifestation of Whipple's disease

Cited by 14 publications

References 13 publications

Whipple’s Disease; An Overlooked Diagnosis

Whipple’s Disease; An Overlooked Diagnosis

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

Chronic, eventually fatal, Kawasaki-like disease in an adult with spondylarthropathy responding to IVIG therapy

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