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

Discussion

We reviewed database cases recorded in PubMed using the following retrieval scheme: ["Whipple disease" and ("infliximab" or "adalimumab" or "etanercept" or "golimumab" or "tocilizumab")]. We gathered the following data from the medical cases reported: age, sex, joint diseases, years with joint disease, TNF-α antagonist therapy, days with TNF-α antagonist therapy before WD was diagnosed, symptoms related to WD, organs affected by WD, investigations for diagnosing WD, treatment and outcome of WD.

We retrieved 14 cases from the PubMed database from January 2004 to December 2014. All the case reports recorded and the two case reports in this manuscript are from European scientists,[4–10] except one case from the United States of America.[11] Four case reports were published in a language other than English.[7,8,10] Table 1 shows age, sex, joint diseases, years with joint disease, TNF-α antagonist therapy, days with TNF-α antagonist therapy before WD was diagnosed, symptoms related to WD, organs affected by WD, investigations for diagnosing WD, treatment and outcome of WD for 16 WD cases. Out of 16 cases, there were 14 men and two women patients with an age range of 33 to 73 years old. Six patients were diagnosed as ankylosis spondylitis (AS), six as seronegative spondyloarthropathy (SA) (seronegative chronic polyarthritis), two as rheumatoid arthritis (RA), one as Still's disease (SD), and one as psoriatic arthritis. All patients had osteoarticular (with pain and swelling) involvement and 15 had gastrointestinal involvement (diarrhea, weight loss, abdominal pain, malabsorption, and so on).[4–12] Moreover, some patients had extra-articular and gastrointestinal involvement, such as vertebra,[5] meningitis,[6] pericarditis (,[6] present report 2 (PR2)), abdominal or thoracic lymph nodes (,[4,6] PR1), gingiva such as scurvy[9] and heart valve involvement.[10] All were diagnosed by histological biopsy, in 14 of 15 cases a PCR assay for DNA detection of T. whipplei was done, and all were positive. The DNA of T. whipplei by PCR assay was detected in duodenal or other gastrointestinal-colonic areas; saliva, blood, feces, bone or cerebrospinal fluid.[4–11] All patients recovered from gastrointestinal involvement when the TNF-α antagonist was stopped and antibiotic treatment was started.

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