Case reportA severe Whipple disease with an immune reconstitution inflammatory syndrome: An additional case of thalidomide efficiency
Section snippets
Discussion
WD is rare, though there is no valid estimate of its actual prevalence. Only about 1000 cases have been reported yet. Although it occurs in people of all ages, the typical patient is a middle-aged white man [3], like our patient. This infectious disease is caused by T. whipplei, first cultured in 2000 [5], which allowed to develop new diagnostic tools as qPCR, and to test in vitro antibiotics susceptibility [4]. Diagnosis relies on histological involvement of small-bowel biopsy (PAS staining
Disclosure of interest
The authors declare that they have no conflicts of interest concerning this article.
Acknowledgements
The authors thank Mrs Valerie Macioce for her support in linguistic adjustment, and H. Lepidi for Fig. 1b.
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2016, Joint Bone SpineCitation Excerpt :Thus, low steroid doses were apparently unable to prevent IRIS onset in the anti-TNF-associated TB context. Other anti-inflammatory treatments, e.g., anti-TNF reintroduction mainly for life-threatening symptoms [15,16] or thalidomide [17,18], have been used. One patient received rituximab for paradoxical lymph-node fistula and active rheumatism, despite high-dose steroids: this regimen allowed rapid steroid dose tapering without further IRIS symptoms.
Immune reconstitution inflammatory syndrome during treatment of Whipple's disease
2015, Joint Bone SpineCitation Excerpt :Oral corticosteroids (0.5–2 mg/kg daily) are generally used with success [5], although they should be reserved for severe or life-threatening disease because of their harmful side effects and so as not to compromise control of the infection. In case of corticosteroid resistance, thalidomide was found to produce a rapid response in two cases [7,10], as also observed in erythema nodosum leprosum. In our patient, treatment with thalidomide was not indicated for the moment because the outcome was favorable.
Atypical focal forms of Whipple's disease seen by rheumatologists
2015, Joint Bone SpineCitation Excerpt :According to a study of heart valve specimens from 1135 patients with infective endocarditis [14], T. whipplei is the fourth most common organism (after streptococci, staphylococci, and enterococci) and the most common cause of infective endocarditis with negative blood cultures. In conclusion, in patients with osteoarticular manifestations, T. whipplei infection should be considered among the possible diagnoses even in the absence of gastrointestinal symptoms [2,4–6,13,15,16], given the severity of this condition. Particular attention to the possibility of T. whipplei infection is warranted in a middle-aged man with intermittent joint symptoms that run an atypical course, without erosions but with persistent systemic inflammation or leukocytosis.
Atypical focal forms of Whipple's disease seen by rheumatologists
2014, Revue du Rhumatisme (Edition Francaise)