Cases reported "Whipple Disease"

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1/18. Whipple's arthritis: direct detection of tropheryma whippelii in synovial fluid and tissue.

    We describe 2 patients presenting with polyarthritis in whom the synovial fluid (1 patient) or synovial tissue (1 patient) was positive for tropheryma whippelii, the Whipple's disease-associated bacillus, when examined by polymerase chain reaction (PCR) and dna sequencing. Histopathologic findings were consistent with articular Whipple's disease in the synovial fluid of 1 patient and the synovial tissue of the other. In both patients, bowel mucosal specimens were negative for Whipple's disease features by histologic and PCR methods. One patient was positive for T whippelii in the peripheral blood. Control synovial fluid specimens from 40 patients with other arthritides, including Lyme arthritis, were negative. Sequencing of a 284-basepair region of the 16S ribosomal rna gene confirmed that the sequence is closely related to the known T whippelii sequence. Both patients responded to treatment with antibiotics.
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2/18. Clinical relapse in Whipple's disease despite maintenance therapy.

    Whipple's disease is a multisystem disorder that was first reported just over 100 years ago. Only recently, the bacillus responsible for the condition was identified and subsequently cultured. However, differences of opinion remain regarding the best antibiotic regimen and duration of therapy at primary diagnosis and there is also great uncertainty about the management of disease relapse. We report a case of clinical relapse of Whipple's disease in a man who was on a prolonged therapy with trimethoprim-sulfamethoxazole. We describe his management and review the literature on the treatment of this condition, with particular reference to the recurrence of the disease.
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3/18. Whipple arthritis: diagnosis by molecular analysis of synovial fluid--current status of diagnosis and therapy.

    Whipple's disease (WD) is an uncommon polysystem infectious disease. In the present report, we describe a patient who presented with a chronic illness consistent with WD and an avascular necrosis of the right hip joint. WD and its proposed causative bacillus, tropheryma whippelii, was identified by molecular analysis (polymerase chain reaction) in bacterial dna extracted from the synovial fluid. The diagnosis was additionally confirmed by upper gastrointestinal endoscopy and a small bowel biopsy with macrophages positive for periodic acid-Schiff reagent demonstrated by light and electron microscopy. This demonstrates that WD can be diagnosed without tissue biopsy. False diagnosis of the polymorphous signs and symptoms of WD can lead to invalidism and even death, whereas correct therapy leads to a cure in most cases. Thus, the current status of diagnosis and therapy is of key importance in the treatment of WD.
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4/18. Whipple's disease with neurological manifestations: case report.

    Whipple's disease (WD) is an uncommon multisystem condition caused by the bacillus tropheryma whipplei. central nervous system involvement is a classical feature of the disease observed in 20 to 40% of the patients. We report the case of a 62 year old man with WD that developed neurological manifestations during its course, and discuss the most usual signs and symptoms focusing on recent diagnostic criteria and novel treatment regimens.
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5/18. Whipple's disease: multiple hospital admissions of a man with diarrhoea, fever and arthralgia.

    Whipple's disease is a rare chronic multi-systemic infectious disorder caused by the Gram-positive bacillus, tropheryma whippelii. Infection may involve any organ in the body, and most commonly affects white men in the fourth to sixth decades of life. The most common presenting symptoms are gastrointestinal and include abdominal pain, diarrhoea, anorexia and associated weight loss. However, the variability in presentation is considerable and some patients may present with intermittent low-grade fever, neurological abnormalities (nystagmus, ophthalmoplegia, cranial nerve defects), migratory arthralgia, lymphadenopathy, or involvement of the cardiovascular system. In typical Whipple's disease, the most severe changes are seen in the proximal small intestine and biopsy reveals mucosal and lymph node infiltration with large, foamy histocytes, containing granules that stain positive with periodic acid-Schiff (PAS) reagent and represent intact or partially degraded bacteria. Extended antibiotic treatment (up to 1-year) is indicated. life-long surveillance for recurrence is essential, once primary treatment has been completed. We report the case of a 58-year-old man who developed a rare infection with the actinobacterium, T. whippelii. The patient had suffered intermittent episodes of varying clinical symptoms associated with multiple hospital admissions and clinical diagnoses, spanning a period of 22 years. Historically, arthralgia was the primary manifestation in this patient and also was the chief complaint for which he was first hospitalized 22 years ago. At his most recent admission to our hospital department, his presenting symptoms were persistent fatigue, weight loss, arthralgia and diarrhoea. Thus, it is essential that clinicians retain a high index of suspicion for T. whippelii infection in patients who have a long-term history of arthritis, fever and diarrhoea.
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6/18. association between Whipple's disease and giardia lamblia infection.

    Whipple' disease is mainly characterized by affecting the digestive system, although it can be a multisystemic process with different clinical symptoms. The bacillus causing the disease has been isolated and cultivated in 2000 and the genome sequence has been recently analyzed in 2003, which means new perspectives for its diagnosis and treatment. giardiasis is an infestation caused by a protozoo and may cause a malabsorption syndrome or run in a subclinic way. The case of a middle-aged male is described, who after a three-year period of migratory arthralgias, showed weight loss, diarrheas and abdominal pain, being diagnosed of giardiasis, and after the persistent symptoms and a number of studies, was diagnosed with whipple disease. Nineteen cases of Giardia-Whipple coinfection have been described in the literature, but the reason of this association has not been found yet. The discussion on whether there is an alteration in the immunitary system which facilitates infections or, the development of an infection lead to the other one, goes on.
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7/18. Whipple's disease can mimic chronic AIDS enteropathy.

    Previous case reports have demonstrated that the intestinal pathology of mycobacterium avium-intracellulare (MAI) infection in the acquired immune deficiency syndrome (AIDS) has a light microscopic appearance similar to Whipple's disease. This case report describes a 52-yr-old male patient with a clinical picture suggestive of AIDS, including diarrhea, weight loss, oral thrush, and intestinal cryptosporidiosis. The intestinal biopsy showed light microscopic features compatible with either MAI or Whipple's disease, but electron microscopy confirmed the presence of the Whipple bacillus. Markers of human immunodeficiency virus (hiv) infection were absent. Although immune abnormalities have been reported in Whipple's disease, this is the first report of opportunistic infections complicating this condition. A useful clinical pearl emerges from this and other cases: AIDS can mimic Whipple's disease; Whipple's disease can mimic AIDS.
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8/18. periodic acid-Schiff-negative granulomatous lymphadenopathy in patients with Whipple's disease. Localization of the Whipple bacillus to noncaseating granulomas by electron microscopy.

    The diagnosis of Whipple's disease in a 58-year-old man was based on the finding of periodic acid-Schiff (PAS)-positive foamy macrophages on duodenal biopsy and demonstration of the typical bacilliform bodies by electron microscopy. The patient also had generalized peripheral lymphadenopathy with lymph node biopsy showing PAS-negative noncaseating granulomas. Electron microscopic examination of the lymph node specimen demonstrated a small number of typical bacilliform bodies with localization specifically to the granulomas in the lymph node. This finding of bacilliform bodies within PAS-negative noncaseating granulomas has not been reported previously. Localization of the Whipple bacillus specifically to noncaseating granulomas suggests that some patients with the disease may manifest a delayed hypersensitivity reaction to the bacillus.
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9/18. Whipple's disease. Demonstration of a persisting monocyte and macrophage dysfunction.

    A patient with Whipple's disease has been followed up for 4 years. Primary involvement was limited to the small intestines, and accumulation of periodic acid-Schiff-positive material, containing typical more or less intact bacillary bodies, was demonstrated within macrophages of affected tissue. After initial oxytetracycline treatment and clinical remission, the patient relapsed, with multiorgan affections. The antibiotic regimen was changed to chloramphenicol, followed by continuous trimethoprim-sulfamethoxazole. Flow cytometric studies showed persisting impairment of monocyte and macrophage intracellular degradation of bacteria during all the 4 years tested. After relapse, reduced activity of several brush border enzymes was demonstrated in distal duodenal biopsy specimens. After 17 months of continuous trimethoprim-sulfamethoxazole therapy complete clinical remission, regression of histopathologic abnormalities, and restoration of duodenal enzyme activities had occurred. The results demonstrate a persisting dysfunction of mononuclear phagocytes from a patient with Whipple's disease, suggesting a primary abnormality of cell-mediated immunity which may promote the susceptibility to the causative bacillus.
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10/18. Electron microscopic detection of Whipple's bacillus in sarcoidlike periodic acid-Schiff-negative granulomas.

    We describe a patient with Whipple's disease without apparent intestinal involvement at initial presentation. Electron microscopy demonstrated the typical bacilli in PAS-negative lymph node and muscle biopsy specimens.
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