Elsevier

Joint Bone Spine

Volume 77, Issue 4, July 2010, Pages 335-339
Joint Bone Spine

Original article
Whipple's disease diagnosed during biological treatment for joint disease

https://doi.org/10.1016/j.jbspin.2010.03.015Get rights and content

Abstract

Objectives

Increased susceptibility to infections is among the main safety concerns raised by biological agents. We describe five cases of Whipple's disease diagnosed during treatment with biological agents.

Methods

We retrospectively identified five cases of Whipple's disease diagnosed between 2003 and 2009 in patients treated with TNFα antagonists in five French hospitals.

Results

Five patients (four male; mean age: 50.4 years; range: 38–67) underwent biological therapy according to prior diagnoses of rheumatoid arthritis (n = 2), ankylosing spondylitis (n = 2), or spondyloarthropathy (n = 1). Biological therapy failed to control the disease, which responded to appropriate antibiotics for Whipple's disease. Retrospectively, clinical symptoms before biological therapy were consistent with Whipple's disease. All five patients had favorable outcomes (mean follow-up, 29 months [13–71]).

Conclusions

Biological therapy probably worsened preexisting Whipple's disease, triggering the visceral disorders. Whipple's disease must be ruled out in patients with joint disease, as patients with this spontaneously fatal condition should not receive immunosuppressive agents.

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:

  • demographics;

  • clinical, laboratory, and imaging study features before TNFα antagonist therapy;

  • 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:

  • migratory intermittent oligoarthritis or polyarthritis involving the hands, wrists and knees, n = 4;

  • inflammatory back or buttock pain, n = 3;

  • relevant morning stiffness, n = 3;

  • intermittent unexplained fever, n = 2;

  • 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.

References (9)

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