Whipple's disease endocarditis following anti-TNF therapy for atypical rheumatoid arthritis

Ansemant, Thiphaine; Célard, M.; Tavernier, C; Maillefert, Jean-Françis; Delahaye, François; Ornetti, Paul

doi:10.1016/j.jbspin.2010.07.003

Cited by 15 publications

(7 citation statements)

References 9 publications

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“…Among the 28 patients for whom data enabled the diagnosis of T. whipplei endocarditis; 16 have been previously reported or cited ( 2 , 5 , 18 , 19 ). According to current modified Duke criteria ( 20 ) before the examination of heart valve specimens, only 1 (3.6%) patient met the criteria for endocarditis and 17 (60.7%) met the criteria for possible endocarditis ( Technical Appendix Table 1 )…”

Section: Resultsmentioning

confidence: 99%

Tropheryma whippleiEndocarditis

Fénollar¹,

Célard²,

Lagier³

et al. 2013

Emerg. Infect. Dis.

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Tropheryma whipplei endocarditis differs from classic Whipple disease, which primarily affects the gastrointestinal system. We diagnosed 28 cases of T. whipplei endocarditis in Marseille, France, and compared them with cases reported in the literature. Specimens were analyzed mostly by molecular and histologic techniques. Duke criteria were ineffective for diagnosis before heart valve analysis. The disease occurred in men 40–80 years of age, of whom 21 (75%) had arthralgia (75%); 9 (32%) had valvular disease and 11 (39%) had fever. Clinical manifestations were predominantly cardiologic. Treatment with doxycycline and hydroxychloroquine for at least 12 months was successful. The cases we diagnosed differed from those reported from Germany, in which arthralgias were less common and previous valve lesions more common. A strong geographic specificity for this disease is found mainly in eastern-central France, Switzerland, and Germany. T. whipplei endocarditis is an emerging clinical entity observed in middle-aged and older men with arthralgia.

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Section: Resultsmentioning

confidence: 99%

Tropheryma whippleiEndocarditis

Fénollar¹,

Célard²,

Lagier³

et al. 2013

Emerg. Infect. Dis.

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“… Data from 19 publications, including 14 case reports, three patient series and one review. DMARD, disease‐modifying anti‐rheumatic drug. …”

Section: Tumour Necrosis Factor Alpha Inhibitors and T Whippleimentioning

confidence: 99%

Systematic review: Whipple's disease (Tropheryma whipplei infection) and its unmasking by tumour necrosis factor inhibitors

Marth

2015

Aliment Pharmacol Ther

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Summary Background The classical form of Whipple's disease (WD), clinically characterised by arthropathy, diarrhoea and weight loss, is rare. Recently, other more frequent forms of Tropheryma whipplei infection have been recognised. The clinical spectrum includes an acute, self‐limiting disease in children, localised forms affecting cardiac valves or the central nervous system without intestinal symptoms, and asymptomatic carriage of T. whipplei which is found in around 4% of Europeans. Genomic analysis has shown that T. whipplei represents a host‐dependent or opportunistic bacterium. It has been reported that the clinical course of T. whipplei infection may be influenced by medical immunosuppression. Aim To identify associations between immunomodulatory treatment and the clinical course of T. whipplei infection. Methods A PubMed literature search was performed and 19 studies reporting on immunosuppression, particularly therapy with tumour necrosis factor inhibitors (TNFI) prior to the diagnosis in 41 patients with Whipple?s disease, were evaluated. Results As arthritis may precede the diagnosis of WD by many years, a relevant percentage (up to 50% in some reports) of patients are treated with immunomodulatory drugs or with TNFI. Many publications report on a complicated Whipple?s disease course or T. whipplei endocarditis following medical immunosuppression, particularly after TNFI. Standard diagnostic tests such as periodic acid–Schiff stain used to diagnose Whipple?s disease often fail in patients who are pre‐treated by TNFI. Conclusions In cases of doubt, Whipple?s disease should be excluded before therapy with TNFI. The fact that immunosuppressive therapy contributes to the progression of T. whipplei infection expands our pathogenetic view of this clinical entity.

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“…Rare Dis. 2009 1 Multisegment spondylitis TNF-a Worsening back pain TNF-a treatment worsened spondylodiscitis, leading to diagnosis of T. Whipplei from rebiopsy of vertebral specimen [ 25 ] Lagier J-C et al Medicine 2010 113 5 of 16 patients with endocarditis as initial symptoms had immunosuppressive treatment Corticosteroids 50 (43 %) TNF-a antagonists 16 (14 %) Others 16 (14 %) Previous immuno suppressive Treatment 56 (50 %) 32 patients (28 %) experienced aggravation of various symptoms after immunosuppressive therapy Patients with inflammatory rheumatoid disease who experience severe general involvment should be screened for T. Whippelii or the therapy is inneffective against polyarthritis [ 26 ] Hoppe E. et al Joint bone Spine 2010 5 RA(2), AS(2), SA(1) TNF a antagonists Failure to control the disease and other symptoms Biological therapy probably worsened pre existing whipples disease [ 27 ] Vancsa A et al Joint Bone Spine 2010 1 Seronegative Oligoarthritis Eternecept Endocarditis DMARDs resistant arthritis should prompt thourough search T.whippelii prior to initiation of TNF-a antagonists [ 28 ] Hmamouci I et al J. Rheumatol. 2010 1 Ankylosing spondylitis Eternecept Scurvy Eternecept probably modified the cytokine environment and thus favoured exercerbation of whipples disease [ 29 ] Daien C.I.…”

Section: Case Presentationmentioning

confidence: 99%

Arthralgia and blood culture-negative endocarditis in middle Age Men suggest tropheryma whipplei infection: report of two cases and review of the literature

et al. 2015

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BackgroundWhipple’s disease is a rare, often multisystemic chronic infectious disease caused by the rod-shaped bacterium Tropheryma whipplei. Very rarely the heart is involved in the process of the disease, leading to culture-negative infective endocarditis. Up to 20 % of all infective endocarditis are blood culture-negative and therefore a diagnostic challenge. We present two unusual cases of culture-negative infective endocarditis encountered in two different patients with prior history of arthralgia. A history of rheumatic arthritis or even a transient arthralgia should put Tropheryma whipplei on the top of differentials in patients of this age group presenting with culture-negative infective endocarditis, especially in cases of therapy resistance to antirheumatic agents.Case presentationThe first patient was a 55 year-old Caucasian male with culture-negative Whipple-related adhesive pericarditis and endocarditis of the aortic valve. Importantly, the patient reported a 15-year history of therapy resistant sero-negative migratory polyarthritis. Aortic valve endocarditis developed during treatment with tocilizumab. The second patient was a 65-year-old male patient with no prior history of the classic Whipple’s disease who presented with a culture-negative aortic valve endocarditis. His past medical history revealed episodes of transient arthralgia, which he was not treated for however, due to the self-limiting nature of the symptoms. Both patients underwent aortic valve replacement surgery. During surgery, pericardectomy was necessary in the first patient due to adhesive pericarditis. Post surgery both patients were started on long-term treatment with trimetoprim-sulfamethoxazol. At 1-year follow-up of both patients, echocardiographic and clinical assessment revealed no signs of persistent infection. Both men reported negative history of arthralgia during the one year period post surgery.ConclusionTropheryma whipplei culture negative-infective endocarditis is an emerging clinical entity, predominantly found in middle-aged and older men with a history of arthralgia. These data highlight the need for ruling out Whipple’s disease in patients with a history of arthralgia prior to initiation of biological agents in treatment of rheumatoid arthritis. There is also a need to assess for Tropheryma whipplei in all patients with culture- negative infective endocarditis.

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Whipple's disease endocarditis following anti-TNF therapy for atypical rheumatoid arthritis

Cited by 15 publications

References 9 publications

Tropheryma whippleiEndocarditis

Tropheryma whippleiEndocarditis

Systematic review: Whipple's disease (Tropheryma whipplei infection) and its unmasking by tumour necrosis factor inhibitors

Arthralgia and blood culture-negative endocarditis in middle Age Men suggest tropheryma whipplei infection: report of two cases and review of the literature

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