Whipple's Disease Diagnosed During Anti-tumor Necrosis Factor Alpha Treatment

Two Case Reports and Review of the Literature

Jose M. Ramos; Francisco Pasquau; Nora Galipienso; Beatriz Valero; Angela Navarro; Agustín Martinez; José Rosas; Ana Gutiérrez; Rosario Sanchez-Martínez

Disclosures

J Med Case Reports. 2015;9(165) 

In This Article

Conclusions

WD is a systemic infection, which involves many chronic manifestations, especially digestive disorders. Some of the manifestations involved are articular symptoms, which can appear as inflammatory rheumatism, such as rheumatoid arthritis or spondylarthritis. Beside these symptoms, the microorganism responsible for T. whipplei disease has also been found in DNA recovered from articular fluid and bones of these patients.[13,14]

Our two cases illustrate that alternative etiologies in patients with articular symptoms should be considered, most importantly following clinical deterioration by immunomodulatory agents like TNF-α inhibitors. The reflections to be made with respect to the analysis of these two cases are to speculate that these patients either had WD with mainly articular manifestations, which had been considered as a rheumatic disease; or in fact, they had a rheumatic disease, which, through the treatment with TNF drugs, may have reactivated T. whipplei and the appearance, therefore, of its florid symptoms that we managed to diagnose.

Therapies with immunomodulators, TNF-α inhibitors, and corticosteroids may transform an infection with T. whipplei, normally at a subacute stage, into a septic, life-threatening disease. Thus, it seems that TNF-α blockade allowed for rapid dissemination of T. whipplei by inhibiting some important immune defense mechanisms. In the later phase of an infection, the TNF-α blocker contributes to limiting the extent of cell and/or tissue damage by inducing apoptosis and maintaining granuloma formation,[15,16] which are important mechanisms in T. whipplei infections. TNF-α blockade might result in the loss of some of the immunological control mechanisms and, therefore, facilitate rapid bacterial dissemination and severe exacerbation of the disease. This is the situation in the first case.

In the second case, the TNF-α blockade was used before the gastrointestinal symptoms appeared. However, with anti-TNF-α treatment, our patient had an acute stroke. After stopping the treatment, our patient recovered from his central nervous system symptoms. When the gastrointestinal involvement of WD was discovered, an examination of DNA for T. whipplei was performed but it was negative. In this case, it was not clear that our patient had neurological involvement of WD, and real improvement of WD after stopping the TNF-α blockade treatment was not clear. There are several cases reported of patients with rheumatic diseases and, during follow-up, they were diagnosed with WD.[13,14,17,18] Thus, the case report of a patient with an initial diagnosis of rheumatoid arthritis who developed pericarditis caused by WD (diagnosed by pericardial biopsy) and the patient who had not been treated previously with TNF-α blocker.[17]

In summary, anti-TNF-α treatment seems to increase the risk of exacerbation of WD and WD may mimic a rheumatic disease.

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