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Subacute infective endocarditis due to Lodderomyces elongisporus: a case report and review of the literature.

ABSTRACT: Lodderomyces elongisporus, a rare emerging pathogen, can cause fungemia often related to immunosuppression or intravenous devices. Herein, we report the case of a 58-year-old woman with subacute infective endocarditis due to Lodderomyces elongisporus identified by blood fungal culture and whole-genome sequencing, who was treated with antifungals, mitral replacement and endocardial vegetation removal surgery.

SUBMITTER: Qiu Y

PROVIDER: S-EPMC10624219 | biostudies-literature | 2023

REPOSITORIES: biostudies-literature

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Subacute infective endocarditis due to Lodderomyces elongisporus: a case report and review of the literature.

Qiu Ye Y Shi Yongying Y Mai Ying Y Wu Zhile Z Guan Jing J Huang Juanni J Su Danhong D Ye Feng F Li Zhengtu Z

Frontiers in public health 20231020

Lodderomyces elongisporus, a rare emerging pathogen, can cause fungemia often related to immunosuppression or intravenous devices. Herein, we report the case of a 58-year-old woman with subacute infective endocarditis due to Lodderomyces elongisporus identified by blood fungal culture and whole-genome sequencing, who was treated with antifungals, mitral replacement and endocardial vegetation removal surgery. ...[more]

PMID: 37927878

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Project description:Infective endocarditis (IE) may be misdiagnosed as ANCA-associated vasculitis (AAV), especially when antineutrophil cytoplasmic antibodies (ANCA) are detected. Distinguishing IE from AAV is crucial to guide therapy. However, little is known about ANCA positivity in IE patients. We present a case report and systematic review of the literature on patients with ANCA-positive IE, aiming to provide a comprehensive overview of this entity and to aid clinicians in their decisions when encountering a similar case. A systematic review of papers on original cases of ANCA-positive IE without a previous diagnosis of AAV was conducted on PubMed in accordance with PRISMA-IPD guidelines. A predefined set of clinical, laboratory, and kidney biopsy findings was extracted for each patient and presented as a narrative and quantitative synthesis. A total of 74 reports describing 181 patients with ANCA-positive IE were included (a total of 182 cases including our own case). ANCA positivity was found in 18-43% of patients with IE. Patients usually presented with subacute IE (73%) and had positive cytoplasmic ANCA-staining or anti-proteinase-3 antibodies (79%). Kidney function was impaired in 72%; kidney biopsy findings were suggestive of immune complexes in 59%, while showing pauci-immune glomerulonephritis in 37%. All were treated with antibiotics; 39% of patients also received immunosuppressants. During follow-up, 69% of patients became ANCA-negative and no diagnosis of systemic vasculitis was reported. This study reviewed the largest series of patients with ANCA-positive IE thus far and shows the overlap in clinical manifestations between IE and AAV. We therefore emphasize that clinicians should be alert to the possibility of an underlying infection when treating a patient with suspected AAV, even when reassured by ANCA positivity. Key Points • This systematic review describes - to our knowledge - the largest series of patients with ANCA-positive infective endocarditis (IE) thus far (N=182), and shows a high degree of overlap in clinical manifestations between IE and ANCA-associated vasculitis (AAV). • ANCA positivity was found in 18-43% of patients with infective endocarditis. Of patients with ANCA-positive IE, the majority (79%) showed cytoplasmic ANCA-staining or anti-PR3-antibodies. We emphasize that clinicians should be alert to the possibility of an underlying infection when treating a patient with suspected AAV, even when reassured by ANCA positivity. • In patients with IE and ANCA-associated symptoms such as acute kidney injury, an important clinical challenge is the initiation of immunosuppressive therapy. All patients with data in this series received antibiotics; 39% also received immunosuppressive therapy. In many of these patients, ANCA-associated symptoms resolved or stabilized after infection was treated. ANCA titers became negative in 69% , and a diagnosis of AAV was made in none of the cases. We therefore recommend that (empiric) antibiotic treatment remains the therapeutic cornerstone for ANCA-positive IE patients, while a watchful wait-and-see approach with respect to immunosuppression is advised.

Project description:Although mycotic aneurysm is a known and important disease in the cerebrovascular system, especially the brain, there are scarce reports about coronary artery mycotic aneurysms (CAMA). CAMA can occur not only in the context of endocarditis but also as a rare adverse event of coronary artery stenting, which has been used more extensively in recent years. Accordingly, it is essential to pay greater attention to its associated presentations and clinical course. Considering the scant evidence available, reporting the disease course of each patient with CAMA can help increase the physician's knowledge about this condition, which is why we are reporting this case. A 42-year-old man with diabetes was referred to our center with embolic left cerebellar infarction 3 months earlier, as well as a 2-month history of feverishness before his referral. His blood culture was positive for Viridans Streptococci, and he had paraclinical signs of inflammation and two- and three-dimensional transthoracic and transesophageal echocardiography (2D & 3D TTE and TEE) signs of aortic and mitral valves' infective endocarditis with the destruction of the aortic valve, severe aortic and mitral regurgitation, severe pulmonary hypertension, and moderate biventricular systolic dysfunction. Regarding the obviously dilated left main coronary artery on TEE images, contrast-enhanced chest multidetector computed tomography was performed for better assessment of coronary arteries with suspicion of CAMA, which confirmed aneurysmal dilatation of the proximal left main coronary artery. The presence of bacteria was confirmed on staining the valvular tissue, resected during the surgical replacement of aortic and mitral valves. As the cardiac surgeon considered CAMA resection and coronary bypass grafting high risk for the patient, he received parenteral antibiotic therapy, for 6 weeks. At 1-year follow-up, he was doing well with no signs/symptoms of endocarditis and well-functioning mechanical prosthetic valves. This case shows the significance of considering CAMA in the setting of endocarditis, resistant to medical and/or surgical therapy or in patients with coronary aneurysm, simultaneous with active endocarditis. Therefore, more attention should be paid to this extravalvular complication of endocarditis, and its possibility should be considered and investigated in any patient presented with valvular endocarditis, especially involving the aortic valve.

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Subacute infective endocarditis due to Lodderomyces elongisporus: a case report and review of the literature.

Publications

Subacute infective endocarditis due to <i>Lodderomyces elongisporus</i>: a case report and review of the literature.

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