Original articleWhipple's disease diagnosed during biological treatment for joint disease☆
Introduction
Increased susceptibility to infections is a major safety concern with biological agents (TNFα antagonists, rituximab, and abatacept). Whipple's disease (WD) is a rare, chronic, systemic infection due to Tropheryma whippleii, a Gram-positive intracellular bacillus related to actinomycetes. WD often manifests as chronic seronegative oligoarthritis or polyarthritis, which may mimic various joint diseases. We describe the cases of five patients who were given biological agents to treat long-standing joint disease (diagnosed as rheumatoid arthritis in two patients, ankylosing spondylitis in two, and spondyloarthropathy in one), then experienced severe organ involvement leading to the diagnosis of WD.
Section snippets
Methods
We identified five cases of WD diagnosed between 2003 and 2009 in patients treated with TNFα antagonists in five French hospitals. TNFα antagonist therapy was initiated according to French Rhumatology Society recommendations for rheumatoid arthritis and ankylosing spondylitis [1], [2]. We abstracted the following data from the medical charts:
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demographics;
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clinical, laboratory, and imaging study features before TNFα antagonist therapy;
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symptoms, investigations, treatment, and outcome of WD.
We
Clinical features before TNFα antagonist therapy
Of the five patients, four were men. Mean age was 50.4 years (38–67) and mean symptom duration was 8.5 years (1–17). Symptoms were as follows:
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migratory intermittent oligoarthritis or polyarthritis involving the hands, wrists and knees, n = 4;
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inflammatory back or buttock pain, n = 3;
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relevant morning stiffness, n = 3;
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intermittent unexplained fever, n = 2;
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night sweats, n = 1.
Retrospectively, mild abdominal symptoms (intermittent pain and/or diarrhea) were noted in three patients (Table 1).
The three patients
Discussion
Five patients given biological agents for long-standing refractory joint disease experienced severe visceral symptoms that led to the diagnosis of WD.
The retrospective review of the patients’ histories showed that most of the clinical symptoms were consistent with WD. Therefore, the biological agents probably caused progression of preexisting WD, triggering the organ involvement. Joint involvement is usually reported in 65 to 90% of patients with WD. Intermittent migratory arthralgia and
Conflict of interest statement
None of the authors has any conflicts of interest to declare.
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The final manuscript has been seen and approved by all the authors and they have given necessary attention to ensure the integrity of the work.