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Whipple's endocarditis: a case report of a blood culture-negative endocarditis.

ABSTRACT: Background:Whipple's disease is caused by Tropheryma whipplei and causes a self-limiting gastrointestinal infection. The majority of the population is an asymptomatic carrier, however, in some patients, it causes an invasive infection with for example arthritis, endocarditis, or involvement of the eyes. Case summary:This case describes a man with long-lasting complaints of progressive dyspnoea caused by heart failure due to total destruction of the aortic and mitral valve as a result of T. whipplei endocarditis, diagnosed with serum polymerase chain reaction. The patient was treated with ceftriaxone and prolonged co-trimoxazole therapy and surgical replacement of the aortic and mitral valve. He was discharged to a rehabilitation centre. Discussion:Tropheryma whipplei is one of the possible microorganisms classified as causing blood culture-negative endocarditis, with predominantly afebrile patients that do not fulfil the Dukes criteria, which makes it difficult to diagnose. Polymerase chain reaction is the cornerstone of the diagnosis. It requires long-term antibiotic treatment up to 12?months. It is recommended by the European Society of Cardiology to discuss treatment in an Endocarditis Team because Whipple's endocarditis has only rarely been described in the literature previously. Whipple's endocarditis has high mortality and relapse rates.

SUBMITTER: Scheurwater MA

PROVIDER: S-EPMC6939820 | biostudies-literature | 2019 Dec

REPOSITORIES: biostudies-literature

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Whipple's endocarditis: a case report of a blood culture-negative endocarditis.

Scheurwater Miriam A MA Verduin Cees M CM van Dantzig Jan-Melle JM

European heart journal. Case reports 20191217 4

<h4>Background</h4>Whipple's disease is caused by <i>Tropheryma whipplei</i> and causes a self-limiting gastrointestinal infection. The majority of the population is an asymptomatic carrier, however, in some patients, it causes an invasive infection with for example arthritis, endocarditis, or involvement of the eyes.<h4>Case summary</h4>This case describes a man with long-lasting complaints of progressive dyspnoea caused by heart failure due to total destruction of the aortic and mitral valve a ...[more]

PMID: 31912002

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Project description:BackgroundCulture-negative endocarditis is uncommon, occurring in less than a third of all cases of infective endocarditis (IE). Culture-negative IE related to a cardiac device is an even greater diagnostic challenge due to its insidious presentation, with onset of symptoms ranging between 3 and 12 months after device implantation. Sensitivity of the modified Duke's criteria remains low in culture-negative and cardiac device-related IE (CDRIE) since classical signs and symptoms of IE are often absent. Small vessel vasculitis has been reported as an immune response to IE. Recognizing immunological phenomenon related to IE is of paramount clinical importance, prompting the search for an underlying infection and avoiding the use of immunosuppressive medications which would otherwise result in an adverse outcome.Case summaryAn 81-year-old Caucasian male presented to the ambulatory medical unit with a two-week history of a symmetrical, generalized purpuric rash. He had an indwelling permanent pacemaker following a transcatheter aortic valve implantation for severe aortic stenosis five years ago. Blood tests showed an iron deficiency anaemia, thrombocytopenia and normal renal function, both CRP and ESR were raised at 61 and 30 mm/hr, respectively. Skin biopsy demonstrated small vessel cutaneous vasculitis. Transthoracic echocardiography revealed a mobile mass measuring 0.9 × 1.7 cm, confirmed on transoesophageal echocardiogram as pacing lead endocarditis. Blood cultures were persistently negative. The patient underwent pacemaker lead extraction, following which the vasculitic rash improved.DiscussionBlood cultures in IE are more likely to be negative if there is a prior antibiotic administration or causative micro-organisms with limited proliferation which fail to grow in conventional media conditions. Transesophageal echocardiography (TOE) offers improved sensitivity and diagnostic yield when compared to transthoracic echocardiography (TTE) in patients with a high clinical suspicion of CDRIE. The evidence in the literature describing culture-negative IE associated with small vessel vasculitis is limited. However, it is recognized that cutaneous small vessel vasculitis may be associated with an underlying bacterial infection. IE produces an inflammatory response, resulting in the deposition of circulating immune complexes and cutaneous signs which are included in the modified Duke's criteria to aid diagnosis. Management of CDRIE requires a multi-disciplinary team approach with an 'Endocarditis Team.' Pacemaker lead infection requires transvenous lead extraction if it is a newly implanted lead. Locking stylets, extraction sheaths or snare retrieval are usually required in cases of older implanted leads. Surgical lead extraction remains the gold standard for larger vegetations (>20 mm) or associated valve endocarditis.

Project description:BackgroundThe prevalence of culture negative infective endocarditis (IEC) is reported as 2-7% though this figure may be as high as 70% in developing countries.1 This higher rate will, at least in part, be due to reduced diagnostic facilities though some data suggests higher rates even when appropriate cultures were taken. The frequency is significantly elevated in patients who have already been exposed to antibiotics prior to blood cultures.1 , 2 A rare cause of culture negative IEC is the HACEK group of organisms that are normal habitants of the oropharyngeal flora and account for 1-3% of native valve endocarditis.3 Aggregatibacter aphrophilus (A. aphrophilus) is a member of the HACEK group of organisms.Case summaryA 32-year-old gentleman with a previous bioprosthetic aortic valve presented with a 1-week history of diarrhoea, vomiting, malaise, and weight loss. He was awaiting redo surgery for stenosis of the bioprosthesis, which had been inserted aged 17 for aortic stenosis secondary to a bicuspid valve. The initial blood tests revealed liver and renal impairment with anaemia. A transoesophageal echocardiogram demonstrated a complex cavitating aortic root abscess, complicated by perforation into the right ventricle. He underwent emergency redo surgery requiring debridement of the aortic abscess, insertion of a mechanical aortic prosthesis (St Jude Medical, USA), annular reconstruction and graft replacement of the ascending aorta. Despite antibiotic therapy, he remained septic with negative blood and tissue cultures. Bacterial 16S rRNA gene sequencing confirmed A. aphrophilus infection, for which intravenous ceftriaxone was initiated. This was subsequently changed to ciprofloxacin due to neutropenia. The patient self-discharged from the hospital during the third week of antibiotic therapy. One week later, he was re-admitted with fever, night sweats, and dyspnoea. Transthoracic echocardiogram revealed a large recurrent aortic abscess cavity around the aortic annulus fistulating into the right heart chambers; this was confirmed by a computed tomography scan. There was dehiscence of the patch repair. Emergency redo aortic root replacement (25 mm mechanical valve conduit, ATS Medical, USA) and annular reconstruction was performed with venoarterial extracorporeal membrane oxygenation (VA-ECMO) support. VA-ECMO was weaned after 3 days. The patient completed a full course of intravenous meropenem and ciprofloxacin and made a good recovery.DiscussionIEC with oropharyngeal HACEK organisms is rare and difficult to diagnose, due to negative blood culture results. The broad-range polymerase chain reaction and gene sequencing with comparison to the DNA database is useful in these circumstances. This case demonstrates the importance of the 16S rRNA gene sequencing for HACEK infection diagnosis and appropriate antibiotic treatment.

Project description:Blood culture-negative endocarditis (BCNE) may represent up to 70% of all endocarditis cases, depending on series. From 2001 to 2009, we implemented in our laboratory a multimodal diagnostic strategy for BCNE that included systematized testing of blood, and when available, valvular biopsy specimens using serological, broad range molecular, and histopathological assays. A causative microorganism was identified in 62.7% of patients.In this study from January 2010 to December 2015, in an effort to increase the number of identified causative microorganisms, we prospectively added to our diagnostic protocol specific real-time (RT) polymerase chain reaction (PCR) assays targeting various endocarditis agents, and applied them to all patients with BCNE admitted to the 4 public hospitals in Marseille, France.A total of 283 patients with BCNE were included in the study. Of these, 177 were classified as having definite endocarditis. Using our new multimodal diagnostic strategy, we identified an etiology in 138 patients (78.0% of cases). Of these, 3 were not infective (2.2%) and 1 was diagnosed as having Mycobacterium bovis BCG endocarditis. By adding specific PCR assays from blood and valvular biopsies, which exhibited a significantly greater sensitivity (P < 10) than other methods, causative agents, mostly enterococci, streptococci, and zoonotic microorganisms, were identified in an additional 27 patients (14 from valves only, 11 from blood only, and 2 from both). Finally, in another 107 patients, a pathogen was detected using serology in 37, valve culture in 8, broad spectrum PCR from valvular biopsies and blood in 19 and 2, respectively, immunohistochemistry from valves in 3, and a combination of several assays in 38.By adding specific RT-PCR assays to our systematic PCR testing of patients with BCNE, we increased the diagnostic efficiency by 24.3%, mostly by detecting enterococci and streptococci that had not been detected by other diagnostic methods, but also agents requiring specific management such as Mycoplasma hominis and Tropheryma whipplei.

Dataset Information

Whipple's endocarditis: a case report of a blood culture-negative endocarditis.

Publications

Whipple's endocarditis: a case report of a blood culture-negative endocarditis.

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