Isolated Pulmonic Valve Endocarditis: Case Report and Review of Existing Literature on Diagnosis and Therapy.
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ABSTRACT:
SUBMITTER: Saleem M
PROVIDER: S-EPMC6833121 | biostudies-other | 2019 Oct
REPOSITORIES: biostudies-other
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Project description:NationaleTrichosporon species are widely distributed in nature and are emerging opportunistic human pathogens. Trichosporon infections are associated with superficial cutaneous involvement in immunocompetent individuals to severe systemic disease in immunocompromised patients. Until now, there is no report in infective endocarditis by Trichosporon mucoides confirmed by molecular diagnostics PATIENT CONCERNS:: A 66-year-old man presented with a fever that had occurred for a period of 6 months. He had undergone aortic valve replacement 10 years prior. Transthoracic echocardiography showed vegetations on the prosthetic aortic valve and native mitral valve. T mucoides was detected in the cultures of blood and vegetations.DiagnosisDNA sequencing using D/D2 region of rRNA and internal transcribed spacer were performed.InterventionsInfections were successfully controlled with valve replacement and voriconazole plus liposomal amphotericin B therapy.OutcomesThere has been no sign of recurrence for 18-months after treatment completion.LessonsThis is the first reported case of infective endocarditis due to T mucoides. Clinicians should consider Trichosporon species as causative agents of endocarditis in patients who have undergone cardiac surgery.
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Project description:Campylobacter fetus is a Gram-negative bacillus typically associated with bacteremia in immunocompromised patients. Prosthetic valve endocarditis (PVE) is a serious complication of prosthetic valve surgery, with a high mortality rate if not treated promptly. We present a rare case of PVE caused by C. fetus. A man in his mid-60s presented to the Emergency Department with a fever and showed elevated C-reactive protein concentrations. He had prosthetic mitral and aortic valve replacement surgery 15 years previously. Gram-negative rods were detected in a blood culture. These rods were identified as C. fetus using matrix-assisted laser desorption ionization time-of-flight mass spectrometry and confirmed by 16S rRNA sequencing. The patient was treated with gentamicin and imipenem, and underwent valve replacement surgery. C. fetus was isolated in a left atrial appendage swab obtained during the surgery. Follow-up blood cultures were negative after treatment. However, after a cardiac arrest event, the patient's general condition deteriorated, and he died. To the best of our knowledge, this is the first case of PVE caused by C. fetus in Korea and the second fatality to date. This case highlights the importance of considering C. fetus as a potential cause of PVE, even in patients without known risk factors.
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Project description:Bullous pemphigoid (BP) is a rare, life-threatening autoimmune blistering disease with pruritus and tension blisters/bullous as the main clinical manifestations. Glucocorticosteroids are the main therapeutic agents for it, but their efficacy is poor in some patients. Tofacitinib, a small molecule agent that inhibits JAK1/3, has shown incredible efficacy in a wide range of autoimmune diseases and maybe a new valuable treatment option for refractory BP. To report a case of refractory BP successfully treated with tofacitinib, then explore the underlying mechanism behind the treatment, and finally review similarities to other cases reported in the literature. Case report and literature review of published cases of successful BP treatment with JAK inhibitors. The case report describes a 73-year-old male with refractory BP that was successfully managed with the combination therapy of tofacitinib and low-dose glucocorticoids for 28 weeks. Immunohistochemistry and RNA sequencing were performed to analyze the underlying mechanism of tofacitinib therapy. A systematic literature search was conducted to identify other cases of treatment with JAK inhibitors. Throughout the 28-week treatment period, the patient experienced clinical, autoantibody and histologic resolution. Immunohistochemical analysis showed tofacitinib significantly decreased the pSTAT3 and pSTAT6 levels in the skin lesions of this patient. RNA sequencing and immunohistochemical testing of lesion samples from other BP patients identified activation of the JAK-STAT signaling pathway. Literature review revealed 17 previously reported cases of BP treated with four kinds of JAK inhibitors successfully, including tofacitinib (10), baricitinib (1), upadacitinib (3) and abrocitinib (3). Our findings support the potential of tofacitinib as a safe and effective treatment option for BP. Larger studies are underway to better understand this efficacy and safety.
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Project description:Lomentospora prolificans (formally Scedosporium prolificans) is an environmental mould with a global distribution. Endocarditis caused by L. prolificans is a rare but serious emerging disease in immunocompromised patients. Prior to this case there have only been eight cases reported in the literature. Diagnosis can be challenging and there are no evidence-based guidelines for treatment.We report a 75-year-old woman with ovarian carcinoma who presented with fever after chemotherapy. Repeated sterile site cultures remained negative until day 22 of admission, when Lomentospora prolificans was isolated from blood cultures. Following extensive investigations, including Fluoro-D-glucose positron emission tomography (FDG-PET) and transoephageal echocardiography (TOE), the patient was diagnosed with endocarditis complicated by cerebral emboli. The patient was considered unsuitable for surgical intervention and passed away five days after the fungus was isolated.Endocarditis caused by Lomentospora prolificans is a rare but emerging condition, with limited treatment options and a high mortality. Awareness of the increasing incidence of Lomentospora prolificans infection, diagnosed often at an advanced stage, with potential for endocarditis may prompt earlier echocardiography or FDG-PET imaging. Further studies are needed to determine the optimal combination and duration of anti-fungal agents, used in conjunction with aggressive surgical excision where feasible.
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Project description:Corynebacterium species are non-fermentous Gram-positive bacilli that are normal flora of human skin and mucous membranes and are commonly isolated in clinical specimens. Non-diphtheriae Corynebacterium are regarded as contaminants when found in blood culture. Currently, Corynebacterium striatum is considered one of the emerging nosocomial agents implicated in endocarditis and serious infections. We report a case of native-valve infective endocarditis caused by C. striatum, which was misidentified by automated identification system but identified accurately by 16S ribosomal RNA sequencing, in a 55-year-old male patient. The patient had two mobile vegetations on his mitral valve, both of which had high embolic risk. Through surgical valve replacement and an antibiotic regimen, the patient recovered completely. In unusual clinical scenarios, C. striatum should not be simply dismissed as a contaminant when isolated from clinical specimens. The possibility of C. striatum infection should be considered even in an immunocompetent patient, and we suggest a genotypic assay, such as 16S rRNA sequencing, to confirm species identity.
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Project description:Reported cases of uni-leaflet mitral valve (MV) were related to the absence or dysplasia of the posterior mitral leaflet with ample anterior mitral leaflet. We present here a new entity of uni-leaflet MV where the MV appears as a membrane-like structure with a single slit-like orifice at its lateral part with no commissures.Case reportContinuous Doppler flow revealed a mean pressure gradient of 19 mmHg across the mitral valve indicating severe mitral stenosis. In 3D images from the left atrial view, the MV appeared like a membrane with a single orifice in its lateral part toward the left atrial appendage, the area of this orifice by 3D was 0.52 cm2, there were no commissures or even any residual lines at the site where commissures should be. The diagnosis of congenital severe mitral stenosis due to acommissural MV was confirmed. During surgery, the surgical appearance of the MV confirmed our diagnosis by 3D.ConclusionIsolated congenital severe mitral stenosis presenting in adulthood is rare, uni-leaflet MV as a cause is only reported in a few cases. MV replacement is usually indicated due to the abnormal anatomy of MV leaflets and the subvalvular apparatus.<Learning objectives: How to diagnose uni-leaflet congenital mitral stenosis and how to differentiate it from other causes of congenital mitral stenosis.>.
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Project description:BackgroundIntestinal Ganglioneuromatosis (IG) is a rare disorder of the enteric nervous system. In pediatric age it is often associated with genetic syndromes such as Neurofibromatosis 1 (NF1), multiple endocrine neoplasia type 2B (MEN2B) and Cowden syndrome (PTEN mutation), and ganglioneuromas (GNs) may be sometimes the first sign of the disease. Isolated GNs are rare and sporadic. Clinical symptom vary and depend on the size and on the location of the GNs. This disorder affects intestinal motility and it, consequently, causes changes in bowel habits, abdominal pain, occlusive symptoms and rarely lower gastrointestinal bleeding secondary to ulceration of the intestinal mucosa. On the other hand, patients can remain asymptomatic for many years.Case presentationWe describe a 9-year-old boy referred to our emergency department for right lower quadrant abdominal pain. No familial history for gastrointestinal disorders. No history of fever or weight loss. At physical examination, he had diffused abdominal pain. Abdominal ultrasonography showed a hypoechoic formation measuring 41.8 mm by 35 mm in the right lower quadrant of the abdomen. Routine blood tests were normal, but fecal occult blood test was positive. Abdominal TC confirmed the hypodense formation, of about 5 cm in transverse diameter, in the right hypochondrium that apparently invaginated in the caecum-last ileal loop. Colonoscopy showed in the cecum an invaginated polypoid lesion of the terminal ileal loop. Laparoscopic resection of the polypoid lesion was performed. Histological diagnosis of the large neoplasm observed in the terminal ileum was diffuse ganglioneuromatosis. NF1, RET and PTEN gene tests resulted negative for specific mutations. At the 1 year follow-up, the patient presented good general condition and blood tests, fecal occult blood test, esophagogastroduodenoscopy, colonoscopy and MR-enterography were negative.ConclusionsOnly few cases are reported in literature of IG in pediatric age. Although rare, the present case suggests that this disorder must be taken in consideration in every patient with GI symptoms such as abdominal pain, constipation, lower intestinal bleeding, in order to avoid a delayed diagnosis.
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