Whipple's Disease in a Patient with Longstanding Seronegative Polyarthritis

Vanderschueren, Dirk; Dequeker, Jan; Geboes, Karel

doi:10.3109/03009748809105282

Cited by 9 publications

(4 citation statements)

References 11 publications

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“…Articular symptoms are the rule rather than the exception in patients with Whipple's disease and thus are the most common extraintestinal manifestations, occurring in up to 90% of patients (Table 2). In about onethird of affected patients (64), these symptoms may precede the gastrointestinal and/or other systemic symptoms by several years, rendering the diagnosis of Whipple's disease difficult (12,21,35,46,59,67,94,106,138,162,165,189). Descriptions of the articular symptoms are often imprecise because they are usually reported only years later upon specific questioning when the diagnosis is finally made.…”

Section: Clinical Manifestationsmentioning

confidence: 99%

Whipple's Disease and “Tropheryma whippelii”

2001

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SUMMARY Whipple's disease is a rare bacterial infection that may involve any organ system in the body. It occurs primarily in Caucasian males older than 40 years. The gastrointestinal tract is the most frequently involved organ, with manifestations such as abdominal pain, malabsorption syndrome with diarrhea, and weight loss. Other signs include low-grade fever, lymphadenopathy, skin hyperpigmentation, endocarditis, pleuritis, seronegative arthritis, uveitis, spondylodiscitis, and neurological manifestations, and these signs may occur in the absence of gastrointestinal manifestations. Due to the wide variability of manifestations, clinical diagnosis is very difficult and is often made only years or even decades after the initial symptoms have appeared. Trimethoprim-sulfamethoxazole for at least 1 year is usually considered adequate to eradicate the infection. The microbiological diagnosis of this insidious disease is rendered difficult by the virtual lack of culture and serodiagnostic methods. It is usually based on the demonstration of periodic acid-Schiff-positive particles in infected tissues and/or the presence of bacteria with an unusual trilaminar cell wall ultrastructure by electron microscopy. Recently, the Whipple bacteria have been characterized at the molecular level by amplification of their 16S rRNA gene(s). Phylogenetic analysis of these sequences revealed a new bacterial species related to the actinomycete branch which was named “Tropheryma whippelli.” Based on its unique 16S ribosomal DNA (rDNA) sequence, species-specific primers were selected for the detection of the organism in clinical specimens by PCR. This technique is currently used as one of the standard methods for establishing the diagnosis of Whipple's disease. Specific and broad-spectrum PCR amplifications mainly but not exclusively from extraintestinal specimens have significantly improved diagnosis, being more sensitive than histopathologic analysis. However, “T. whippelii” DNA has also been found in persons without clinical and histological evidence of Whipple's disease. It is unclear whether these patients are true asymptomatic carriers or whether differences in virulence exist among strains of “T. whippelii” that might account for the variable clinical manifestations. So far, six different “T. whippelii” subtypes have been found by analysis of their 16S-23S rDNA spacer region. Further studies of the pathogen “T. whippelii” as well as the host immune response are needed to fully understand this fascinating disease. The recent cultivation of the organisms is a promising major step in this direction.

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Section: Clinical Manifestationsmentioning

confidence: 99%

Whipple's Disease and “Tropheryma whippelii”

2001

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“…The mean solar time from the event of articular symptoms to the diagnosis is nearly 7 years, 7 but there are some reports focusing the eye on the very late diagnosis of Whipple’s disease, within the absence of gastrointestinal symptoms. 8 , 9 Arthritis has been reported in 41%-61% of cases. 7 All patients had increased CRP, while rheumatoid factor and anti-CCP were absent, and everyone was initially misclassified as suffering from seronegative rheumatoid arthritis.…”

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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“…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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Whipple's Disease in a Patient with Longstanding Seronegative Polyarthritis

Cited by 9 publications

References 11 publications

Whipple's Disease and “Tropheryma whippelii”

Whipple's Disease and “Tropheryma whippelii”

Whipple’s Disease; An Overlooked Diagnosis

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

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