Successful treatment of immune reconstitution inflammatory syndrome in Whipple's disease using thalidomide

Lagier, Jean-Christophe; Fénollar, Florence; Lépidi, Hubert; Liozon, É.; Raoult, Didier

doi:10.1016/j.jinf.2009.09.017

Cited by 45 publications

(20 citation statements)

References 11 publications

(43 reference statements)

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“…If the inflammation does not subside within 24 h, additional or alternative immunosuppressive agents should be prescribed (218,219). Thalomidine seems to be a good alternative treatment in cases of corticosteroid resistance (221,222).…”

Section: Other Treatment Considerationsmentioning

confidence: 99%

Clinical Manifestations, Treatment, and Diagnosis of Tropheryma whipplei Infections

et al. 2017

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SUMMARY Whipple's disease is a rare infectious disease that can be fatal if left untreated. The disease is caused by infection with Tropheryma whipplei, a bacterium that may be more common than was initially assumed. Most patients present with nonspecific symptoms, and as routine cultivation of the bacterium is not feasible, it is difficult to diagnose this infection. On the other hand, due to the generic symptoms, infection with this bacterium is actually quite often in the differential diagnosis. The gold standard for diagnosis used to be periodic acid-Schiff (PAS) staining of duodenal biopsy specimens, but PAS staining has a poor specificity and sensitivity. The development of molecular techniques has resulted in more convenient methods for detecting T. whipplei infections, and this has greatly improved the diagnosis of this often missed infection. In addition, the molecular detection of T. whipplei has resulted in an increase in knowledge about its pathogenicity, and this review gives an overview of the new insights in epidemiology, pathogenesis, clinical manifestations, diagnosis, and treatment of Tropheryma whipplei infections.

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Section: Other Treatment Considerationsmentioning

confidence: 99%

Clinical Manifestations, Treatment, and Diagnosis of Tropheryma whipplei Infections

et al. 2017

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“…The spectrum of chronic infections caused by T. whipplei includes endocarditis, neurological infections, uveitis, arthritis and osteoarticular infections and, less frequently, isolated adenopathies [2,6]. Moreover, as a result of this culture, the recommended treatment is no longer empirical but evidence-based, on the grounds of both in vitro results and clinical outcomes [7,8]. This includes a 1-year combination of doxycycline and hydroxychloroquine followed by a lifetime treatment with doxycycline [7,[9][10] because of the lifelong susceptibility of Whipple's disease patients to this bacterium [9].…”

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confidence: 99%

Acute Infections Caused by Tropheryma Whipplei

2017

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Tropheryma whipplei is the causative bacterium of Whipple's disease. Its first culture has led to an enlargement of the field of the caused infections. Here, we comprehensively review acute T. whipplei infections. In a cohort study featuring 4000 children, T. whipplei was significantly more common in patients with diarrhea (4%) than in those without (1.7%). A case–controlled study highlighted 58 patients suffering from pneumonia with the detection of T. whipplei in their bronchoalveolar fluids. Finally, a recent study detected T. whipplei in the blood of 36 febrile patients experiencing pulmonary symptoms in a rural area of Senegal. T. whipplei is definitively an agent of acute gastroenteritis, a cause of nonmalarial fever in Africa, and probably a cause of pulmonary infections.

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“…A minor role for pathogen specificity and a more important impact of dysregulated innate immune reactions in IRIS pathogenesis have been discussed very recently (34). This perspective is supported by the fact that immunogenetic factors involved in Ag presentation influence the susceptibility to CWD IRIS (35), and that IRIS in CWD has been successfully treated with thalidomide (36), supposedly by downregulating TNF-a expression.…”

Section: Discussionmentioning

confidence: 96%

Immunopathology of Immune Reconstitution Inflammatory Syndrome in Whipple’s Disease

Moos

Feurle

Schinnerling

et al. 2013

The Journal of Immunology

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During antimicrobial treatment of classic Whipple’s disease (CWD), the chronic systemic infection with Tropheryma whipplei, immune reconstitution inflammatory syndrome (IRIS), is a serious complication. The aim of our study was to characterize the immunological processes underlying IRIS in CWD. Following the definition of IRIS, we describe histological features of IRIS and immunological parameters of 24 CWD IRIS patients, 189 CWD patients without IRIS, and 89 healthy individuals. T cell reconstitution, Th1 reactivity, and the phenotype of T cells were described in the peripheral blood, and infiltration of CD4+ T cells and regulatory T cells in the duodenal mucosa was determined. During IRIS, tissues were heavily infiltrated by CD3+, predominantly CD45RO+CD4+ T cells. In the periphery, initial reduction of CD4+ cell counts and their reconstitution on treatment was more pronounced in CWD patients with IRIS than in those without IRIS. The ratio of activated and regulatory CD4+ T cells, nonspecific Th1 reactivity, and the proportion of naive among CD4+ T cells was high, whereas serum IL-10 was low during IRIS. T. whipplei–specific Th1 reactivity remained suppressed before and after emergence of IRIS. The findings that IRIS in CWD mainly are mediated by nonspecific activation of CD4+ T cells and that it is not sufficiently counterbalanced by regulatory T cells indicate that flare-up of pathogen-specific immunoreactivity is not instrumental in the pathogenesis of IRIS in CWD.

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Successful treatment of immune reconstitution inflammatory syndrome in Whipple's disease using thalidomide

Cited by 45 publications

References 11 publications

Clinical Manifestations, Treatment, and Diagnosis of Tropheryma whipplei Infections

Clinical Manifestations, Treatment, and Diagnosis of Tropheryma whipplei Infections

Acute Infections Caused by Tropheryma Whipplei

Immunopathology of Immune Reconstitution Inflammatory Syndrome in Whipple’s Disease

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