scholarly journals Whipple’s disease scleral nodules: a novel presentation in 2 consecutive patients

2020 ◽  
Vol 20 (1) ◽  
Author(s):  
Waleed K. Alsarhani ◽  
Muhannad I. Alkhalifah ◽  
Hind M. Alkatan ◽  
Afaf L. Alsolami ◽  
Azza M. Y. Maktabi ◽  
...  
Keyword(s):  
Systemic Disease ◽  
Periodic Acid ◽  
Surgical Excision ◽  
Successful Outcome ◽  
Whipple’S Disease ◽  
Whipple's Disease ◽  
Periodic Acid Schiff ◽  
Systemic Involvement ◽  
Male Farmer ◽  
Systemic Symptoms

Abstract Background Whipple’s disease (WD) is a rare, chronic, infection caused by gram-positive filamentous aerobic actinobacterium Tropheryma whipplei occurs classically in the gastrointestinal tract and shows histopathologically foamy macrophages with typical numerous PAS-positive, non-acid fast particles. Ocular WD in the form of uveitis may occur in the absence of systemic disease but has not been reported to present with scleral manifestation. We describe for the first time to the best of our knowledge 2 cases of scleral nodules with typical histopathological morphology of WD and without systemic involvement. Case presentation The first was a 53-year old diabetic male farmer who presented with 2 nontender right eye scleral nodules for 3 months, had a negative systemic workup, and surgical excision showed Periodic acid Schiff (PAS)-positive eosinophilic structures inside macrophages. Grocott’s methenamine silver (GMS) stain and acid-fast bacilli (AFB) stain of the tissue itself were negative. The second case was a 60-year old male who presented with an asymptomatic superior scleral nodule for 4 months, which showed similar appearance and negative GMS and AFB stains. Conclusion WD should be included in the differential diagnosis of scleral nodules even in the absence of systemic symptoms. Surgical excision without systemic treatment resulted in successful outcome without recurrence.

scholarly journals Whipple’s Disease: Our Own Experience and Review of the Literature

2013 ◽  
Vol 2013 ◽  
pp. 1-10 ◽  
Author(s):  
Jan Bureš ◽  
Marcela Kopáčová ◽  
Tomáš Douda ◽  
Jolana Bártová ◽  
Jan Tomš ◽  
...  
Keyword(s):  
Systemic Disease ◽  
Periodic Acid ◽  
Favourable Outcome ◽  
Penicillin G ◽  
Low Grade ◽  
Whipple’S Disease ◽  
Review Of The Literature ◽  
Intestinal Histology ◽  
Whipple's Disease ◽  
Periodic Acid Schiff

Whipple’s disease is a chronic infectious systemic disease caused by the bacteriumTropheryma whipplei. Nondeforming arthritis is frequently an initial complaint. Gastrointestinal and general symptoms include marked diarrhoea (with serious malabsorption), abdominal pain, prominent weight loss, and low-grade fever. Possible neurologic symptoms (up to 20%) might be associated with worse prognosis. Diagnosis is based on the clinical picture and small intestinal histology revealing foamy macrophages containing periodic-acid-Schiff- (PAS-) positive material. Long-term (up to one year) antibiotic therapy provides a favourable outcome in the vast majority of cases. This paper provides review of the literature and an analysis of our 5 patients recorded within a 20-year period at a tertiary gastroenterology centre. Patients were treated using i.v. penicillin G or amoxicillin-clavulanic acid + i.v. gentamicin for two weeks, followed by p.o. doxycycline (100 mg per day) plus p.o. salazopyrine (3 g per day) for 1 year. Full remission was achieved in all our patients.

scholarly journals Tropheryma whipplei Detection by Nanopore Sequencing in Patients With Interstitial Lung Disease

2021 ◽  
Vol 12 ◽  
Author(s):  
Yifan Guo ◽  
Lijuan Li ◽  
Zhenzhong Li ◽  
Lingxiao Sun ◽  
Hui Wang
Keyword(s):  
Interstitial Lung Disease ◽  
Lung Disease ◽  
Quantitative Polymerase Chain Reaction ◽  
Periodic Acid ◽  
Tropheryma Whipplei ◽  
Whipple’S Disease ◽  
Nanopore Sequencing ◽  
Whipple's Disease ◽  
Periodic Acid Schiff ◽  
Laboratory Findings

Tropheryma whipplei is a bacterium associated with Whipple’s disease, which commonly manifests as weight loss, arthralgia, and diarrhea. The most frequently involved organs comprise the heart and eyes, in addition to the central nervous system. Few studies have explored the relationship between T. whipplei and pneumonia. Herein, we report three patients with interstitial lung disease (ILD) of unknown cause, whose bronchoalveolar lavage fluid (BALF) were evaluated via Nanopore sequencing. In our in-house BALF Nanopore platform, human DNA was removed with saponin, to improve the reads ratio of microorganisms/host. T. whipplei was the sole or most abundant pathogen in all the patients, comprising 1,385, 826, and 285 reads. The positive result was confirmed via quantitative polymerase chain reaction (PCR) with two pairs of primers (cycle threshold value: 33.26/36.29; 31.68/32.01; 28.82/28.80) and Sanger sequencing. To our knowledge, this is the first report of T. whipplei detection using Nanopore-based sequencing. The turnaround time was approximately 6–8 h in clinical laboratories, including less than 1 h for analysis. In conclusion, the results of this study confirm that Nanopore sequencing can rapidly detect rare pathogens, to improve clinical diagnosis. In addition, diagnosis of Whipple’s disease should be combined other laboratory findings, such as periodic acid-Schiff (PAS) staining, and considered a possibility in middle-aged men presenting with ILD and a clinical history of unexplained arthralgia and/or fever.

Periodic acid-schiff-negative granulomatous lymphadenopathy in patient with whipple's disease

1987 ◽  
Vol 83 (1) ◽  
pp. 165-170 ◽  
Author(s):  
Gilbert M. Wilcox ◽  
Bruce S. Tronic ◽  
Diane J. Schecter ◽  
Martin J. Arron ◽  
David F. Righi ◽  
...  
Keyword(s):  
Periodic Acid ◽  
Whipple’S Disease ◽  
Whipple's Disease ◽  
Periodic Acid Schiff

scholarly journals Role of enteroscopy in the diagnosis of whipple’s disease

2021 ◽  
Vol 84 (4) ◽  
pp. 665-667
Author(s):  
M Mascarenhas Saraiva ◽  
T Ribeiro ◽  
T Costa Pereira ◽  
P Andrade ◽  
H Cardoso ◽  
...  
Keyword(s):  
Periodic Acid ◽  
Clinical Manifestations ◽  
Systemic Infection ◽  
Whipple’S Disease ◽  
Upper Endoscopy ◽  
Whipple's Disease ◽  
Periodic Acid Schiff ◽  
Abdominal Symptoms ◽  
Duodenal Biopsies

Whipple’s disease is a rare chronic systemic infection caused by Tropheryma whipplei. The widespread infection by this rod is responsible for the protean clinical manifestations of the disease, although its classical form is notable for the prevalence of abdominal symptoms such as chronic diarrhea and abdominal pain. Whitish-yellow patches, suggestive of lymphangiectasia, are typically observed in the duodenum during upper endoscopy. The diagnosis of this condition is supported by the identification in duodenal biopsies of Periodic acid-Schiff staining within lamina propria macrophages. Nevertheless, a significant portion of patients do not have lesions within the range of conventional upper endoscopy. Therefore, other endoscopic procedures such as video capsule endoscopy and enteroscopy may be useful to detect more distal lesions. The authors describe a case where the combined used of both techniques allowed the unmasking of this disease.

scholarly journals Whipple’s Disease Affecting Ileal Peyer’s Patches: The First Case Report

2019 ◽  
Vol 2019 ◽  
pp. 1-4
Author(s):  
Sabah Sid’Amar ◽  
Giacomo Puppa
Keyword(s):  
Lymphoid Tissue ◽  
Diagnostic Yield ◽  
Clinical Investigation ◽  
Periodic Acid ◽  
Disease Diagnosis ◽  
Peyer's Patches ◽  
Peyer’S Patches ◽  
Whipple’S Disease ◽  
Whipple's Disease ◽  
First Case

Whipple’s disease is a rare chronic systemic bacterial infectious disease which can affect multiple organs, with a wide clinical spectrum encompassing many symptoms presenting in various forms and combinations. In the cases where the gastrointestinal tract is implicated, the more frequent localizations involve the small bowel, especially the duodenum. A case of a 67-year-old man who underwent clinical investigation after presenting with a progressive weight loss and showing a hypercapting right paracoeliac adenopathy at PET-CT scan is reported herein. A gastroscopy and a colonoscopy were done. The biopsies of the endoscopically normal ileal mucosa encompassed some submucosal Peyer’s patches. Histological examination of this lymphoid tissue revealed several foamy macrophages which turned out positive on periodic acid-Schiff special staining. Polymerase chain reaction of the microdissected lymph follicles allowed for confirming Whipple’s disease diagnosis. A targeted antibiotic treatment administrated to the patient led to a rapid clinical improvement. This finding of a previously unreported localization of infected macrophages in Whipple’s disease suggests that sampling the organized mucosal-submucosal lymphoid tissue may increase the diagnostic yield in endoscopic biopsies.

scholarly journals Whipple's Disease and “Tropheryma whippelii”

2001 ◽  
Vol 14 (3) ◽  
pp. 561-583 ◽  
Author(s):  
Fabrizio Dutly ◽  
Martin Altwegg
Keyword(s):  
Bacterial Species ◽  
Periodic Acid ◽  
Clinical Manifestations ◽  
The Body ◽  
Rrna Gene ◽  
Low Grade ◽  
Whipple’S Disease ◽  
Major Step ◽  
Whipple's Disease ◽  
Initial Symptoms

SUMMARY Whipple's disease is a rare bacterial infection that may involve any organ system in the body. It occurs primarily in Caucasian males older than 40 years. The gastrointestinal tract is the most frequently involved organ, with manifestations such as abdominal pain, malabsorption syndrome with diarrhea, and weight loss. Other signs include low-grade fever, lymphadenopathy, skin hyperpigmentation, endocarditis, pleuritis, seronegative arthritis, uveitis, spondylodiscitis, and neurological manifestations, and these signs may occur in the absence of gastrointestinal manifestations. Due to the wide variability of manifestations, clinical diagnosis is very difficult and is often made only years or even decades after the initial symptoms have appeared. Trimethoprim-sulfamethoxazole for at least 1 year is usually considered adequate to eradicate the infection. The microbiological diagnosis of this insidious disease is rendered difficult by the virtual lack of culture and serodiagnostic methods. It is usually based on the demonstration of periodic acid-Schiff-positive particles in infected tissues and/or the presence of bacteria with an unusual trilaminar cell wall ultrastructure by electron microscopy. Recently, the Whipple bacteria have been characterized at the molecular level by amplification of their 16S rRNA gene(s). Phylogenetic analysis of these sequences revealed a new bacterial species related to the actinomycete branch which was named “Tropheryma whippelli.” Based on its unique 16S ribosomal DNA (rDNA) sequence, species-specific primers were selected for the detection of the organism in clinical specimens by PCR. This technique is currently used as one of the standard methods for establishing the diagnosis of Whipple's disease. Specific and broad-spectrum PCR amplifications mainly but not exclusively from extraintestinal specimens have significantly improved diagnosis, being more sensitive than histopathologic analysis. However, “T. whippelii” DNA has also been found in persons without clinical and histological evidence of Whipple's disease. It is unclear whether these patients are true asymptomatic carriers or whether differences in virulence exist among strains of “T. whippelii” that might account for the variable clinical manifestations. So far, six different “T. whippelii” subtypes have been found by analysis of their 16S-23S rDNA spacer region. Further studies of the pathogen “T. whippelii” as well as the host immune response are needed to fully understand this fascinating disease. The recent cultivation of the organisms is a promising major step in this direction.

scholarly journals Isolated Knee Arthritis as Early and Only Symptom of Whipple’s Disease

2018 ◽  
Vol 2018 ◽  
pp. 1-4 ◽  
Author(s):  
Dario Giunchi ◽  
Natalie Marcoli ◽  
Luca Deabate ◽  
Marco Delcogliano ◽  
Enrique Testa ◽  
...  
Keyword(s):  
Gastrointestinal Tract ◽  
Scientific Literature ◽  
Systemic Disease ◽  
Whipple’S Disease ◽  
Whipple's Disease ◽  
Knee Arthritis ◽  
Specific Data ◽  
Pas Staining ◽  
Inflammatory Infiltrates ◽  
Disease Specialist

We report a case of isolated Whipple’s disease involving the knee of a 64-year-old female patient who presented recurrent monoarthritis whose origin was not clear. Initially, the cause of the gradually invalidating symptoms was related to a meniscal lesion and a diffuse minor grade chondropathy, but pain and functional impairment suggested that more exams were needed. Biopsies were performed during arthroscopy. The histology showed highly inflammatory infiltrates with PAS staining negative for Tropheryma while PCR revealed the infection with Tropheryma whipplei. This, following the recommendation of a rheumatologist and infectious disease specialist, led to biopsies of the gastrointestinal tract and analysis of the cerebrospinal fluid that showed no other organ involvement. This confirms the scientific literature that an isolated monoarthritis without involvement of the gastrointestinal tract caused by this bacterium is rare but can occur as an early manifestation of potentially fatal systemic disease. Moreover, a review of the scientific literature showed the uncertainty about epidemiology of this rare disease, suggesting that more and specific data are required.

scholarly journals Juvenile Xanthogranuloma Presenting as Bilateral Non-Infiltrative Extraconal Superior Orbital Tumour in a 27 Year Old Nigerian Woman: Features, Management and Outcome

2012 ◽  
Vol 5 ◽  
pp. CCRep.S10138 ◽  
Author(s):  
Okosa Michael Chuka ◽  
Anyiam Daniel Chukwuemeka Darlinton
Keyword(s):  
Systemic Disease ◽  
Rare Condition ◽  
Surgical Excision ◽  
Juvenile Xanthogranuloma ◽  
Case Presentation ◽  
Nigerian Woman ◽  
Upper Lid ◽  
Systemic Manifestations ◽  
Systemic Symptoms

Introduction Orbital masses in adults are often caused by systemic diseases or are associated with systemic manifestations. Juvenile xanthogranuloma as a cause is rare and unreported in Africa. We present clinical features, management, and outcomes of bilateral orbital adult onset juvenile xanthogranuloma. Case Presentation A 27 year old Nigerian woman presented with bilateral upper-lid lumps having lasted 5 months. These increased in size for about 1 month and stopped. Lid swelling was preceded by itchy eyes, redness of conjunctiva, and occasional mild pain. There were no visual or systemic symptoms. The lumps were firm, slightly mobile, not tender, and not attached to skin but rather to deeper structures. There was restriction on up-gaze but no proptosis or diplopia. Hematological, biochemical, and X-ray investigations were normal. Prednisolone tablets 10 mg daily for two weeks were not useful. Tissue biopsy was invaluable in diagnosis of this rare condition and disclosed juvenile xanthogranuloma. Partial surgical excision was done under lidocaine infiltration. No recurrence has occurred in 40 months of follow-up. No systemic disease has manifested. Conclusion Juvenile xanthogranuloma can present as bilateral superior orbital tumor in adults; functional and cosmetic aims were achieved by sub-total excision.

scholarly journals Whipple's disease. Report of five cases with different clinical features

2001 ◽  
Vol 43 (1) ◽  
pp. 45-50 ◽  
Author(s):  
Maria de Lourdes de Abreu FERRARI ◽  
Eduardo Garcia VILELA ◽  
Luciana Costa FARIA ◽  
Claudia Alves COUTO ◽  
Célio Jefferson SALGADO ◽  
...  
Keyword(s):  
Clinical Manifestation ◽  
Systemic Disease ◽  
Light And Electron Microscopy ◽  
Clinical Manifestations ◽  
Ocular Involvement ◽  
Jejunal Mucosa ◽  
Whipple’S Disease ◽  
Whipple's Disease ◽  
Full Remission

Whipple's disease (WD) is a rare systemic disease of infectious etiology which involves the small intestine but can virtually affect any organ. We present here five cases (four males and one female) ranging in age from 20 to 59 years. All patients had intestinal involvement associated or not with clinical manifestations linked to this organ. Vegetation in the tricuspid valve was observed in one patient, suggesting endocarditis caused by Tropheryma whippelii, with disappearance of the echocardiographic alterations after treatment. In one of the male patients the initial clinical manifestation was serologically negative spondylitis, with no diarrhea occurring at any time during follow-up. Ocular involvement associated with intestinal malabsorption and significant weight loss were observed in one case. In the other two cases, diarrhea was the major clinical manifestation. All patients were diagnosed by histological examination of the jejunal mucosa and, when indicated, of extraintestinal tissues by light and electron microscopy. After antibiotic treatment, full remission of symptoms occurred in all cases. A control examination of the intestinal mucosa performed after twelve months of treatment with sulfamethoxazole-trimethoprim revealed the disappearance of T. whippelii in four patients. The remaining patient was lost to follow-up.

scholarly journals Gastric B-Cell Lymphoma with Mott Cell Differentiation in a Dog

2009 ◽  
Vol 21 (5) ◽  
pp. 715-719 ◽  
Author(s):  
Gabrita De Zan ◽  
Valentina Zappulli ◽  
Laura Cavicchioli ◽  
Linda Di Martino ◽  
Eriberta Ros ◽  
...  
Keyword(s):  
Cell Differentiation ◽  
B Cell ◽  
Gastrointestinal Disease ◽  
Periodic Acid ◽  
Palliative Therapy ◽  
Clinical Signs ◽  
B Cell Lymphoma ◽  
Surgical Excision ◽  
Periodic Acid Schiff ◽  
Regional Lymph Nodes

A gastric lymphoid tumor with involvement of regional lymph nodes and spleen was diagnosed in an 8-year-old crossbreed male dog with a 6-month history of gastrointestinal disease. Despite surgical excision and palliative therapy (prednisolone and cimetidine), the dog was euthanized due to worsening of clinical signs. At necropsy, multiple white, solid, nodular, infiltrative masses were observed in the stomach, duodenum, spleen, liver, and lungs in association with generalized lymph node enlargement. Cytology, histology, histochemistry, immunohistochemistry, and electron microscopy revealed that the neoplastic cell population was composed of B lymphocytes that contained variable amounts of round periodic acid-Schiff-positive cytoplasmic globules consistent with Russell bodies. The tumor most likely represented a variant of B-cell neoplasia with extensive Mott cell differentiation.

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