scholarly journals Rare but Not so Rare? The Evolving Spectrum of Whipple's Disease

2001 ◽  
Vol 12 (3) ◽  
pp. 133-135 ◽  
Author(s):  
John M Conly ◽  
B Lynn Johnston
Keyword(s):  
Case Reports ◽  
Case Series ◽  
Individual Case ◽  
Pulmonary Involvement ◽  
Pcr Analysis ◽  
Whipple’S Disease ◽  
Whipple's Disease ◽  
Regional Lymph Nodes ◽  
First Case ◽  
Gradual Loss

Knowledge about Whipple's disease began to emerge in 1907, when George Hoyt Whipple recognized the first case of the disease that now bears his name. He reported the case of a 36-year-old physician with "a gradual loss of weight and strength, stools consisting chiefly of neutral fat and fatty acids, indefinite abdominal signs, and a peculiar multiple arthritis" (1). Findings at autopsy consisted of poly-serositis, aortic valve vegetations and deposition of fat in the intestinal mucosa and regional lymph nodes with marked infiltration by foamy macrophages (1). It was originally thought to be a disorder of fat metabolism, and the term 'intestinal lipodystrophy' was proposed. Whipple's disease has since been recognized as a rare, multivisceral, chronic disease with a clinical presentation dominated by a symptom triad of diarrhea, weight loss and malabsorption. However, digestive symptoms are often preceded for months or years by other symptoms, the most common being arthralgia, although cardiovascular, neurological or pulmonary involvement may be more prominent at times. Once considered the ideal case report, recent characterization ofTropheryma whippeliiby means of broad range bacterial ribosomal DNA polymerase chain reaction (PCR) analysis (2,3) and its subsequent cultivation (4) has led to a veritable explosion of individual case reports, case series and hitherto unrecognized manifestations of the disease, such that it is now considered an underdiagnosed infectious disease (5). It is timely to provide an update on new developments in Whipple's disease.

scholarly journals Different Manifestations of Rare Cases of Unruptured Sinus of Valsalva Aneurysm—Case Series and Literature Review

2018 ◽  
Vol 28 (03) ◽  
pp. 202-206 ◽  
Author(s):  
Bhagya Narayan Pandit ◽  
Siva Subramaniyan ◽  
Tarun Kumar ◽  
Richa Agrawal ◽  
Deepankar Vatsa
Keyword(s):  
Case Reports ◽  
Case Series ◽  
Individual Case ◽  
Sinus Of Valsalva ◽  
Aortic Sinus ◽  
Sinus Of Valsalva Aneurysm ◽  
Severe Aortic Regurgitation ◽  
First Case ◽  
Threatening Condition ◽  
Life Threatening

AbstractSinus of Valsalva aneurysm (SVA) is rare, and aneurysm of the left sinus of Valsalva is extremely rare cardiovascular disease. Clinical presentation can vary from mostly asymptomatic to catastrophic life-threatening emergency. We report four extremely rare cases of SVA with different manifestations of which one case involved left aortic sinus with large unruptured aneurysm causing severe mitral regurgitation (MR) and severe aortic regurgitation (AR). The second case aneurysm was from right sinus presented with trifascicular block with intermittent complete heart block (CHB). Third was a diagnosed case of unruptured right SVA and while awaiting for surgery he had sudden cardiac death (SCD) at home. Therefore, SVA can present from asymptomatic to life-threatening condition, such as SCD. To the best of our knowledge, there are individual case reports in literature, and this is the first case series of unruptured SVA in literature highlighting the rarity of this disease.

Herpetic Whitlow of the Hand in Infants

2020 ◽  
Author(s):  
Mohammad M. Al-Qattan ◽  
Nada G. AlQadri ◽  
Ghada AlHayaza
Keyword(s):  
Bacterial Infection ◽  
Case Reports ◽  
Case Series ◽  
Individual Case ◽  
Senior Author ◽  
Secondary Bacterial Infection ◽  
Clinical Appearance ◽  
History Of ◽  
Mode Of Transmission ◽  
Time Of Presentation

Abstract Introduction Herpetic whitlows in infants are rare. Previous authors only reported individual case reports. We present a case series of six infants. Materials and Methods This is a retrospective study of six cases of herpetic whitlows in infants seen by the senior author (MMA) over the past 23 years (1995–2017 inclusive). The following data were collected: age, sex, digit involved in the hand, mode of transmission, time of presentation to the author, clinical appearance, presence of secondary bacterial infection, presence of other lesions outside the hand, method of diagnosis, treatment, and outcome. Results All six infants initially presented with classic multiple vesicles of the digital pulp. In all cases, there was a history of active herpes labialis in the mother. Incision and drainage or deroofing of the vesicles (for diagnostic purposes) resulted in secondary bacterial infection. Conclusion The current report is the first series in the literature on herpetic whitlows in infants. We stress on the mode of transmission (from the mother) and establishing the diagnosis clinically. In these cases, no need for obtaining viral cultures or polymerase chain reaction; and no medications are required. Once the vesicles are disrupted, secondary bacterial infection is frequent and a combination of oral acyclovir and intravenous antibiotics will be required.

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.

Abstract P101: Mechanical Thrombectomy in COVID-19 Positive Patients With Large Vessel Occlusion: Pooled Review of Individual Case Series

Stroke ◽  
2021 ◽  
Vol 52 (Suppl_1) ◽  
Author(s):  
Muhammad Z Memon ◽  
Taha Nisar ◽  
Amit Singla ◽  
Anil Nanda ◽  
Gaurav Gupta ◽  
...  
Keyword(s):  
Clinical Outcomes ◽  
Mechanical Thrombectomy ◽  
Case Reports ◽  
Case Series ◽  
Individual Case ◽  
Large Vessel ◽  
Large Vessel Occlusion ◽  
Vessel Occlusion ◽  
Historic Data ◽  
Meta Analyses

Background: COVID-19 has been shown to induce a hypercoagulable state thereby increasing the risk of arterial thrombosis resulting in Large Vessel Occlusion Stroke (LVOs) Objective: We performed a systematic review of published reports to study the clinical characteristics, and outcomes of COVID-19 acute ischemic stroke (AIS) patients with LVO treated with mechanical thrombectomy (MT) and compared them with historical controls. Methods: We conducted a systematic literature search from December 2019 to July 2020 using multiple combinations of keywords from PubMed and Ovid databases according to the PRISMA meta-analyses and systemic reviews guidelines and then pooled data from individual case series. We included studies where COVID -19 associated LVO cases were treated with MT and their clinical outcomes were reported. We then compared these findings with the historic patient data from the five landmark randomized MT trials, the Hermes collaborators (HC). Results: An initial search generated 12 studies but after excluding case reports and multiple reports comprising of the same series of patients, a total of five reports consisting of 51 patients were analyzed. The mean age of patients was 59 years (IQR 36-75), and 40 (78 %) were men. Median NIHSS on presentation was 20 (IQR 10-29). AIS with LVO was the presenting manifestation of COVID-19 in 16 (20%) of patients. Intracranial ICA was the most common site of occlusion found in 27 (53%) of patients with multi-territory occlusion in 10 (20 %). Final recanalization TICI ≥ 2b was achieved in 33 (64%) of patients but reocclusion was noted in 7 (14 %). Modified Rankin score (mRS) 0-2 was reported in 12 (23 %) of patients with 40 % in-hospital mortality. When compared to historic data from HC, COVID -19 patients were younger (59 vs 69 years), presented with a higher median NIHSS score (20 vs 17), and had a higher prevalence of ICA terminus occlusion (53% vs 21% ). Similarly, patient outcomes were poor in the COVID -19 group with mRs 0-2 in (23 % versus 46 %) and mortality (40 % vs 15 %) compared to Hermes group. Conclusion: COVID -19 AIS patients with LVO who underwent MT were younger, had multiple territory occlusions with a propensity for ICA terminus location, and had poor angiographic and clinical outcomes as compared to historic data.

scholarly journals B.04 Distal and asymmetric myasthenia gravis: a case series of 54 patients

2016 ◽  
Vol 43 (S2) ◽  
pp. S10-S10
Author(s):  
AG Florendo-Cumbermack ◽  
MW Nicolle
Keyword(s):  
Myasthenia Gravis ◽  
Clinical Course ◽  
Case Reports ◽  
Delayed Diagnosis ◽  
Case Series ◽  
Retrospective Chart Review ◽  
Individual Case ◽  
Response To Treatment ◽  
Antibody Testing ◽  
Repetitive Nerve Stimulation

Background: Distal/asymmetric presentations of myasthenia gravis (MG) are uncommon and occur in 3-7% of patients with MG. This pattern of weakness is often not recognized as a manifestation of MG, leading to inappropriate investigations, delayed diagnosis and potentially missed opportunities for treatment. Our knowledge about this atypical presentation is limited to small case series and individual case reports. This study therefore aims to expand our understanding by describing the clinical course, diagnosis and treatment of a larger series of patients with this presentation. Methods: We conducted a retrospective chart review of patients with definite MG (either acetylcholine receptor [AChR] or MuSK antibody positive or clear evidence of postsynaptic neuromuscular junction dysfunction on electrodiagnostic studies), who attended the MG Clinic in London. Details of the clinical course, electrodiagnostic studies, antibody testing and response to treatment are reported. Results: 5.9% (54/921) of patients with definite MG had distal/asymmetric limb involvement, 56% at onset and 4% developing more than 10 years later. Males predominated (2:1). Finger extensors were most affected. 83% were AChR antibody positive. 7% had thymomas. On repetitive nerve stimulation most patients showed the most significant decrement distally on the more affected side. Almost all patients improved with treatment. Conclusions: This study expands our understanding of distal/asymmetric presentations of MG.

scholarly journals Prophylaxis of Invasive Fungal Infections: A Review of the Use of Posaconazole

2009 ◽  
Vol 1 ◽  
pp. CMT.S1948
Author(s):  
Curtis D. Collins ◽  
Jeannina A. Smith ◽  
Daniel R. Kaul
Keyword(s):  
At Risk ◽  
Fungal Pathogens ◽  
Fungal Infections ◽  
Case Reports ◽  
Case Series ◽  
Individual Case ◽  
Invasive Fungal Infections ◽  
Cost Of Care ◽  
Therapeutic Drug ◽  
Patients At Risk

Invasive fungal infections (IFIs) cause significant morbidity, mortality, and increased cost of care in patients with hematological malignancies, prolonged (i.e. >7-10 days) treatment induced neutropenia, and other disease states causing underlying immunosuppression. One strategy often used to combat the development of invasive infections is the use of antifungal agents as prophylaxis in at risk patients. Posaconazole is an oral triazole with a useful spectrum of activity against many fungal pathogens of concern in patients at risk for the development of IFIs. Posaconazole is only available in oral formulation and therapeutic drug monitoring may provide value due to variable absorption and serum concentrations. Clinical efficacy and pharmacoeconomic data have demonstrated the utility of posaconazole in the treatment of oropharyngeal candidiasis and for prophylaxis in patients at risk for development of IFIs. Several organizations or expert groups involved in developing guidelines for the management of IFIs recommend posaconazole anti-fungal prophylaxis in patients with AML or MDS and chemotherapy induced neutropenia or significant GVHD. In addition, nonrandomized studies (largely of salvage therapy) and case series suggest that posaconazole may be effective as treatment for invasive aspergillosis, zygomycosis, and coccidiomycosis. Further, small case series or individual case reports suggest activity against other less commonly encountered filamentous fungi and Histoplasma.

scholarly journals Reversible Pulmonary Hypertension Associated with Whipple’s Disease

2012 ◽  
Vol 2012 ◽  
pp. 1-2
Author(s):  
A. Villa ◽  
G. Nucera ◽  
A. Kostihova ◽  
A. Mazzola ◽  
P. Marino
Keyword(s):  
Pulmonary Hypertension ◽  
Antibiotic Therapy ◽  
Respiratory Symptoms ◽  
Systolic Pressure ◽  
Pulmonary Involvement ◽  
Pulmonary Artery Systolic Pressure ◽  
Whipple’S Disease ◽  
Whipple's Disease ◽  
History Of ◽  
Possible Cause

We describe a case of Whipple’s disease with pulmonary hypertension in a 72-year-old woman in whom the pulmonary hypertension resolved completely after antibiotic therapy. She was admitted to study with a 2-months history of weight loss, diarrhoea, abdominal pain, asthenia, inappetence, and fever. She did not have dyspnoea or respiratory symptoms. A casual echocardiogram showed a pulmonary artery systolic pressure of 95 mmHg. Forty days after starting antibiotic therapy, an echocardiogram showed a complete normalisation of right ventricular involvement. Whipple’s disease is a rare and multisystemic disorder in which pulmonary involvement is not a well-known finding. Although Whipple’s disease is not generally considered as a possible cause of pulmonary hypertension, such awareness is important because it may be potentially resolved with antibiotic therapy.

Endovascular intervention for acute stroke due to infective endocarditis

2012 ◽  
Vol 32 (2) ◽  
pp. E1 ◽  
Author(s):  
Haitham Dababneh ◽  
V. Shushrutha Hedna ◽  
Jenna Ford ◽  
Ziad Taimeh ◽  
Keith Peters ◽  
...  
Keyword(s):  
Infective Endocarditis ◽  
Acute Stroke ◽  
Case Reports ◽  
Case Series ◽  
Standard Of Care ◽  
Clinical Decision ◽  
Neurological Complications ◽  
Endovascular Interventions ◽  
First Case ◽  
Risk Of Hemorrhage

The overall incidence of neurological complications due to infective endocarditis is as high as 40%, with embolic infarcts more common than hemorrhagic strokes. The standard of care for typical strokes does not apply to infective endocarditis because there is a substantial risk of hemorrhage with thrombolysis. In the last decade there have been multiple case reports of intravenous and intraarterial thrombolysis with successful outcomes for acute strokes with related infective endocarditis, but successful endovascular interventions for acute strokes associated with infective endocarditis are rarely reported. To the authors' knowledge, this report is the first case in the literature to use a mechanical retrieval device in successful vegetation retrieval in an infective endocarditis acute stroke. Although an interventional approach for treatment of acute stroke related to infective endocarditis is a promising option, it is controversial and a cautious clinical decision should be made on a case-by-case basis. The authors conclude that this approach can be tested in a case series with matched controls, because this condition is rare and a randomized clinical trial is not a realistic option.

scholarly journals Prenatal Diagnosis of Neuroblastoma

2014 ◽  
Vol 8 (3) ◽  
pp. 321-327
Author(s):  
Mona Zvanca ◽  
Cristian Andrei
Keyword(s):  
Prenatal Diagnosis ◽  
Case Reports ◽  
Case Series ◽  
Expectant Management ◽  
Individual Case ◽  
Management Options ◽  
Tertiary Center ◽  
Low Incidence ◽  
Tumor In Childhood

ABSTRACT Fetal malignancies are rare complications during pregnancies, but when they appear, they are very challenging for the perinatology team. Because of their low incidence, the information is limited, with data provided from individual case reports or small case series. Although neuroblastoma is the most frequent extracranial solid tumor in childhood, prenatal diagnosis by ultrasound is very rare and almost always discovered during routine third trimester ultrasound. Expectant management is usually indicated prenatally, with serial ultrasound examination. Delivery should be planned in a tertiary center together with pediatric oncologists and surgeons to allow appropriate postnatal management. We present two cases of neuroblastoma diagnosed at 36 and 33 weeks of gestation with multiple aspects of this tumor identified by ultrasound. Both cases needed surgery and had a favorable outcome. The key role of ultrasound in diagnosis and follow-up of neuroblastoma in pregnancy is discussed, together with the management options recommended in literature. How to cite this article Andrei C, Vladareanu R, Zvanca M, Vladareanu S. Prenatal Diagnosis of Neuroblastoma. Donald School J Ultrasound Obstet Gynecol 2014;8(3):321-327.

scholarly journals Emergence of evidence during disease outbreaks: lessons learnt from the Zika virus outbreak

2020 ◽  
Author(s):  
Michel J Counotte ◽  
Kaspar W Meili ◽  
Nicola Low
Keyword(s):  
Infectious Diseases ◽  
Study Design ◽  
Zika Virus ◽  
Case Reports ◽  
Emerging Infectious Diseases ◽  
Case Series ◽  
Disease Outbreaks ◽  
Basic Research ◽  
First Case ◽  
Over Time

AbstractIntroductionOutbreaks of infectious diseases trigger an increase in scientific research and output. Early in outbreaks, evidence is scarce, but it accumulates rapidly. We are continuously facing new disease outbreaks, including the new coronavirus (SARS-nCoV-2) in December 2019.The objective of this study was to describe the accumulation of evidence during the 2013-2016 Zika virus (ZIKV) outbreak in the Pacific and the Americas related to aetiological causal questions about congenital abnormalities and Guillain-Barré syndrome.MethodsWe hypothesised that the temporal sequence would follow a pre-specified order, according to study design. We assessed 1) how long it takes before findings from a specific study design appear, 2) how publication of preprints could reduce the time to publication and 3) how time to publication evolves over time.ResultsWe included 346 publications published between March 6, 2014 and January 1, 2019. In the 2013-–2016 ZIKV outbreak, case reports, case series and basic research studies were published first. Case-control and cohort studies appeared between 400–700 days after ZIKV was first detected in the region of the study origin. Delay due to the publication process were lowest at the beginning of the outbreak. Only 4.6% of the publications was available as preprints.DiscussionThe accumulation of evidence over time in new causal problems generally followed a hierarchy. Preprints reduced the delay to initial publication. Our methods can be applied to new emerging infectious diseases.

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