Previous immunosuppressive therapy is a risk factor for immune reconstitution inflammatory syndrome in Whipple's disease

Biagi, Federico; Trotta, Lucia; M, Di Stefano; Balduzzi, Davide; Marchese, A.; Vattiato, C.; Bianchi, Paola Ilaria; Fénollar, Florence; Corazza, Gino Roberto

doi:10.1016/j.dld.2012.05.008

Cited by 34 publications

(23 citation statements)

References 21 publications

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“…Oral corticosteroids are the first-choice treatment, and patients usually respond rapidly. 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%

“…Biological treatment with TNF-␣ inhibitors has been shown to be fatal due to exacerbation with a mean time of 26 months after the start of treatment (107,217). Previous immunosuppressive treatment is a major risk factor for the development of immune reconstitution inflammatory syndrome (IRIS) in classic Whipple's disease (218,219).…”

Section: Other Treatment Considerationsmentioning

confidence: 99%

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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%

Section: Other Treatment Considerationsmentioning

confidence: 99%

Clinical Manifestations, Treatment, and Diagnosis of Tropheryma whipplei Infections

et al. 2017

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“…This condition is characterized by a severe general inflammatory status with fever and arthralgias occurring few days after antibiotic treatment . In our patient, previous intake of immunosuppressive drug for arthritis could have represented a risk factor for immune‐reconstitution inflammatory syndrome development .…”

Section: Discussionmentioning

confidence: 79%

Transmission electron microscopy helpfulness in Whipple's disease masked by immunosuppressant therapy for arthritis

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Losurdo

Iannone

et al. 2017

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A 61-year-old woman received a diagnosis of undifferentiated non-erosive arthritis in 2010 and assumed methotrexate and steroids in 2014. After 1 year, she experienced watery diarrhea, vomiting, fever, weight loss, and severe hypoalbuminemia, thus being admitted into our Unit. Esophagogastroduodenoscopy showed duodenal lymphangiectasia and duodenal biopsy samples several foamy PAS-positive macrophages and villous subtotal atrophy. Transmission electron microscope demonstrated several extracellular and intracellular rod-shaped bacteria (Tropheryma whipplei). Therefore, we diagnosed Whipple's disease. Our patient assumed doxycycline/hydroxychloroquine with prompt remission of gastrointestinal symptoms. At 1 year of follow-up, she was symptom-free, histological reassessment showed almost complete mucosal healing and transmission electron microscope demonstrated bacteria breaking/disappearance. The present report demonstrates that: (i) rheumatological manifestations are common onset symptoms of Whipple's disease; (ii) immunosuppressive therapy may delay the diagnosis and worsen clinical presentation; (iii) transmission electron microscopy for specific bacteria detection/disappearance is an helpful diagnostic tool, when available.

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“…In a large proportion of WD-associated IRIS cases, however, patients had complete resolution of the symptoms before a late recurrence of inflammation, in contrast to this case [18]. IRIS has been described as a recognised complication of WD with a rate of up to 10%, and is sometimes interpreted as undertreatment or recurrent disease [18,19].…”

Section: Discussionmentioning

confidence: 99%

Bilateral Ocular Myositis Associated with Whipple's Disease

et al. 2016

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Purpose: To describe the clinical features of a Caucasian female patient with a history of treated gastrointestinal Whipple's disease (WD) who developed new-onset diplopia, with a description of the histopathological features of the extraocular muscle biopsies. Methods: A previously fit 38-year-old Caucasian female presented with acute-onset diplopia after being on a sustained medication regime for biopsy-proven gastrointestinal WD. A magnetic resonance imaging scan of her orbits with gadolinium revealed diffuse enhancement of the bellies of the extraocular muscles bilaterally, particularly the medial rectus, superior rectus, and superior oblique muscles, consistent with an infiltrative myositis. She underwent unilateral extraocular muscle biopsies. Results: The extraocular muscle biopsies contained macrophages between the muscle fibres. These contained periodic acid-Schiff-positive cytoplasmic granules. Immunohistochemistry with an antibody raised to Tropheryma whipplei showed positive staining of the same macrophages. Transmission electron microscopy confirmed the presence of effete T. whipplei cell membranes in lysosomes. Conclusion: This case describes bilateral WD-associated extraocular muscle myositis. The exact mechanism for this unusual presentation is unclear, but both a WD-associated immune reconstitution inflammatory syndrome and treatment failure are possibilities, with a good response observed to antibiotic therapy and adjunctive corticosteroids.

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Previous immunosuppressive therapy is a risk factor for immune reconstitution inflammatory syndrome in Whipple's disease

Cited by 34 publications

References 21 publications

Clinical Manifestations, Treatment, and Diagnosis of Tropheryma whipplei Infections

Clinical Manifestations, Treatment, and Diagnosis of Tropheryma whipplei Infections

Transmission electron microscopy helpfulness in Whipple's disease masked by immunosuppressant therapy for arthritis

Bilateral Ocular Myositis Associated with Whipple's Disease

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