Cutaneous Granulomatosis Revealing Whipple’s Disease: Value of Tropheryma whipplei Polymerase Chain Reaction Assay for the Diagnosis

Whipple’s Disease is a rare systemic infectious disease caused by the ubiquitous actinomycetes Tropheryma whipplei (T. whipplei). We report herein a rare case of a cutaneous granulo matosis with hypercalcemia as an unusual presenting feature of Whipple’s disease. The diagnosis of the bacteria was obtained from skin and inguinal lymph node biopsy (16 rDNA PCR screening and histological examination using PAS staining). T. whipplei was also identified on saliva and stool specimens, using specific PCR and colonic biopsies. Treatment with hydroxychloroquine and doxycycline allowed a rapid resolution of symptoms with a complete recovery.

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Can Whipple's Disease Be Transmitted by Gastroscopes?

Infection Control and Hospital Epidemiology ◽

10.1086/502188 ◽

2003 ◽

Vol 24 (3) ◽

pp. 191-194 ◽

Cited By ~ 15

Author(s):

Bernard La Scola ◽

Jean-Marc Rolain ◽

Max Maurin ◽

Didier Raoult

Keyword(s):

Polymerase Chain Reaction ◽

Bactericidal Activity ◽

False Positive ◽

Tropheryma Whipplei ◽

Whipple’S Disease ◽

Chain Reaction ◽

Whipple's Disease ◽

Cell Monolayers ◽

Polymerase Chain ◽

Positive Results

AbstractObjective:To determine whether disinfection protocols currently used for gastroscopes are effective against cultures ofTropheryma whipplei.Design:The bactericidal activity of 2% glutaraldehyde and two peracetic acids on the Twist-Marseille strain ofT. whippleigrown in cell monolayers was determined.Patients:Two patients who were diagnosed as having Whipple's disease 3 years after they had had intestinal biopsies.Results:The disinfectants reduced bacteria by approximately 2 log10to 3 log10after 5 to 60 minutes of contact.Conclusion:The bactericidal activity of a disinfectant is usually considered significant if it causes a 5 log10or greater reduction in viable bacterial titers. Disinfecting gastroscopes with2%glutaraldehyde or peracetic acids for 20 minutes may be insufficient to prevent transmission ofT. whippleion the instruments or stop false-positive results on polymerase chain reaction.

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Tropheryma whipplei, Immunosuppression and Whipple's Disease: From a Low-Pathogenic, Environmental Infectious Organism to a Rare, Multifaceted Inflammatory Complex

Digestive Diseases ◽

10.1159/000369538 ◽

2015 ◽

Vol 33 (2) ◽

pp. 190-199 ◽

Cited By ~ 10

Author(s):

Thomas Marth

Keyword(s):

Heart Valves ◽

Clinical Manifestations ◽

Systemic Infection ◽

Tropheryma Whipplei ◽

Molecular Techniques ◽

Whipple’S Disease ◽

Whipple's Disease ◽

New Findings ◽

The Central Nervous System ◽

Body Compartments

Background: The actinobacterium Tropheryma whipplei was detected 20 years ago by molecular techniques, and following its culture has been characterized as the cause of a systemic infection known as Whipple's disease (WD). T. whipplei occurs in the environment, is prevalent only in humans, is believed to be transmitted via oral routes and to be host dependent. Key Messages: The classical form of T. whipplei infection, i.e. classical WD (CWD), is rare. It is well defined as slowly progressing chronic infection with arthralgia, diarrhea and weight loss, mostly in middle-aged men. However, current research revealed a much broader spectrum of clinical features associated with T. whipplei infection. Thus, T. whipplei may cause acute and transient infections (observed primarily in children) and the bacterium, which is found in soil and water, occurs in asymptomatic carriers as well as in CWD patients in clinical remission. In addition, T. whipplei affects isolated and localized body compartments such as heart valves or the central nervous system. Subtle immune defects and HLA associations have been described. New findings indicate that the progression of asymptomatic T. whipplei infection to clinical WD may be associated with medical immunosuppression and with immunomodulatory conditions. This explains that there is a discrepancy between the widespread occurrence of T. whipplei and the rareness of WD, and that T. whipplei infection triggered by immunosuppression presents with protean clinical manifestations. Conclusions: This review highlights recent findings and the clinical spectrum of infection with T. whipplei and WD, focusing specifically on the role of host immunity and immunosuppression. Current concepts of the pathogenesis, diagnosis and therapy are discussed.

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Tropheryma whipplei, the Whipple's disease bacillus, induces macrophage apoptosis through the extrinsic pathway

Cell Death and Disease ◽

10.1038/cddis.2010.11 ◽

2010 ◽

Vol 1 (4) ◽

pp. e34-e34 ◽

Cited By ~ 21

Author(s):

L Gorvel ◽

K Al Moussawi ◽

E Ghigo ◽

C Capo ◽

J-L Mege ◽

...

Keyword(s):

Tropheryma Whipplei ◽

Whipple’S Disease ◽

Extrinsic Pathway ◽

Whipple's Disease ◽

Macrophage Apoptosis

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Whipple's disease: rare disorder and late diagnosis

Revista do Instituto de Medicina Tropical de São Paulo ◽

10.1590/s0036-46652012000500010 ◽

2012 ◽

Vol 54 (5) ◽

pp. 293-297 ◽

Cited By ~ 5

Author(s):

Viviane Plasse Renon ◽

Marcelo Campos Appel-da-Silva ◽

Rafael Bergesch D'Incao ◽

Rodrigo Mayer Lul ◽

Luciana Schmidt Kirschnick ◽

...

Keyword(s):

Gastrointestinal Symptoms ◽

Disease Diagnosis ◽

Endoscopic Biopsy ◽

Upper Gastrointestinal Endoscopy ◽

Whipple’S Disease ◽

Whipple's Disease ◽

Polymerase Chain ◽

One Year ◽

Infectious Disorder

Whipple's disease is a rare systemic infectious disorder caused by the bacterium Tropheryma whipplei. We report the case of a 61-year-old male patient who presented to emergency room complaining of asthenia, arthralgia, anorexia, articular complaints intermittent diarrhea, and a 10-kg weight loss in one year. Laboratory tests showed the following results: Hb = 7.5 g/dL, albumin = 2.5 mg/dL, weight = 50.3 kg (BMI 17.4 kg/m²). Upper gastrointestinal endoscopy revealed areas of focal enanthema in the duodenum. An endoscopic biopsy was suggestive of Whipple's disease. Diagnosis was confirmed based on a positive serum polymerase chain reaction. Treatment was initiated with intravenous ceftriaxone followed by oral trimethoprim-sulfamethoxazole. After one year of treatment, the patient was asymptomatic, with Hb = 13.5 g/dL, serum albumin = 5.3 mg/dL, and weight = 70 kg (BMI 24.2 kg/m²). Whipple's disease should be considered a differential diagnosis in patients with prolonged constitutional and/or gastrointestinal symptoms. Appropriate antibiotic treatment improves the quality of life of patients.

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Eosinophilic vasculitis: A Rare Presentation of Whipple’s Disease

Canadian Journal of Gastroenterology ◽

10.1155/2007/326174 ◽

2007 ◽

Vol 21 (3) ◽

pp. 189-191 ◽

Cited By ~ 10

Author(s):

Waleed Al-hamoudi ◽

Fadi Habbab ◽

Carmine Nudo ◽

Ayoub Nahal ◽

Kenneth Flegel

Keyword(s):

Lymph Node ◽

Lymph Node Involvement ◽

Lymph Node Biopsy ◽

Whipple’S Disease ◽

Whipple's Disease ◽

Rare Presentation ◽

Negative Results ◽

Mild Anemia ◽

Node Involvement ◽

Foamy Macrophages

Whipple’s disease is a multisystem infectious disease caused by the bacteriumTropheryma whippelii. A case with an unusual presentation is reported. A 66-year-old man presented with a febrile vasculitic rash on his forearms. An extensive rheumatological, hematological and infectious workup gave negative results, apart from mild anemia and eosinophilia. An abdominal computed tomography revealed a retroperitoneal lymphadenopathy, and a skin biopsy revealed an eosinophilic vasculitis. This diverted the work toward ruling out a lymphoma or a vasculitic process. A lymph node biopsy was then performed and showed a diffuse neutrophilic inflammation with abundant foamy macrophages, fat necrosis and lipogranuloma formation. These findings were considered to be nonspecific and no further pathological investigation was carried out. After a course of corticosteroids, diarrhea and weight loss predominated and subsequently a diagnosis of Whipple’s disease was confirmed on a small-bowel biopsy. Lymph node involvement was then confirmed on re-evaluation using the appropriate stains.

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Meningo-Ependymitis in Whipple's Disease

Canadian Journal of Neurological Sciences / Journal Canadien des Sciences Neurologiques ◽

10.1017/s0317167100023556 ◽

1980 ◽

Vol 7 (2) ◽

pp. 163-167 ◽

Cited By ~ 15

Author(s):

L.R. Lapointe ◽

J. Lamarche ◽

A. Salloum ◽

R. Beaudry

Keyword(s):

Complete Recovery ◽

Whipple’S Disease ◽

Gait Ataxia ◽

Whipple's Disease ◽

Inflammatory Infiltration ◽

Asymptomatic Patients ◽

Elevated Protein ◽

Cerebral Involvement ◽

Inflammatory Changes ◽

Infectious Disorder

SUMMARY:Six years after apparent complete recovery from intestinal Whipple's disease, a 56 year old man developed insidious progressive somnolence and gait ataxia. Studies showed hydrocephalus with obstruction of the aqueduct and CSF leukocytosis and elevated protein. Arachnoid biopsy during craniotomy revealed chronic inflammatory infiltration with PAS-positive macrophages. The patient died 5 years later despite two courses of antibiotic therapy. This is the first report of histologically confirmed cerebral Whippie's disease during life.Whipple's disease is a systemic infectious disorder. Cerebral involvement even in neurologically asymptomatic patients should be sought with periodic CSF cytologic studies and a search for hydrocephalus. The possibility of cerebral Whipple's disease should be considered in the presence of unexplained hydrocephalus and/or chronic inflammatory changes in the spinal fluid, especially in those with past or active intestinal disease.

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Tropheryma whippleiCrystalline Keratopathy: Report of a Case and Updated Review of the Literature

Case Reports in Ophthalmological Medicine ◽

10.1155/2012/707898 ◽

2012 ◽

Vol 2012 ◽

pp. 1-7 ◽

Cited By ~ 3

Author(s):

Scott D. Schoenberger ◽

Sumeer Thinda ◽

Stephen J. Kim

Keyword(s):

Signs And Symptoms ◽

Tropheryma Whipplei ◽

Extensive Literature ◽

Whipple’S Disease ◽

Whipple's Disease ◽

Case Presentation ◽

Search Terms ◽

Crystalline Keratopathy ◽

Extensive Literature Search ◽

English Articles

Purpose. To report a case ofTropheryma whippleiinfection with crystalline keratopathy and review the recent literature on the presentation, diagnosis, and management of Whipple's disease.Methods. Detailed case presentation and extensive literature search of Pubmed for all years through February 2012 using the following search terms:Whipple's disease, Tropheryma whipplei, corneal deposits, crystalline keratopathy, and uveitis. Relevant articles were retrieved and analyzed. English abstracts were used for non-English articles. Cross-referencing was employed and reference lists from selected articles were used to identify additional pertinent articles.Results. Diagnosis of Whipple's disease remains challenging and untreated infection can result in mortality. Ocular signs and symptoms are usually nonspecific, but several independent cases have reported the presence of intraocular crystals or crystalline-like deposits.Conclusions. The presence of intraocular crystals or crystalline-like deposits may be an identifying feature of ocular Whipple’s disease.

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