HHV-6 associated fulminant encephalo-myocarditis in an immunocompetent individual.
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ABSTRACT:
SUBMITTER: Maramattom BV
PROVIDER: S-EPMC4683890 | biostudies-literature | 2015 Oct-Dec
REPOSITORIES: biostudies-literature
Publications
HHV-6 associated fulminant encephalo-myocarditis in an immunocompetent individual.
Annals of Indian Academy of Neurology 20151001 4
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Project description:BackgroundROS1 tyrosine kinase inhibitors are one of the primary immunotherapies for ROS1 fusion-positive cancers. Tyrosine kinase inhibitors have markedly improved outcomes for advanced cancers previously with poor prognosis. Entrectinib is an example of an ROS1 inhibitor that can be used for lung adenocarcinoma. There are numerous adverse effects with rare cardiac side effects reported, such as heart failure and myocarditis.Case summaryA 27-year-old male being treated for lung adenocarcinoma who presented new congestive heart failure 2 weeks after starting Entrectinib. He developed refractory ventricular tachycardia, cardiogenic shock with an endomyocardial biopsy that showed active lymphohistiocytic myocarditis. With antiarrhythmic therapy, heavy sedation, mechanical circulatory support, and high-dose steroids, the patient had complete resolution of symptoms and return to baseline status.DiscussionThis is a rare case with a severe complication after starting Entrectinib for lung adenocarcinoma. In the literature, this is the first case of its kind presenting with myocarditis and severe heart failure after treatment with Entrectinib. This case highlights not only using cardiac imaging, and biopsy to help guide the diagnosis, but also describe the appropriate management.
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Project description:Fatal myocarditis is a rare complication in immunosuppressed children. Recent reports have linked human herpesvirus 6 (HHV-6) infection, typically a benign infection in childhood, with myocarditis. HHV-6 can reactivate during periods of immunosuppression. Here, we report 2 cases in which children were immunosuppressed, one for treatment of Evans syndrome and the other post hematopoietic stem cell transplantation, who developed rapid and fatal HHV-6-associated myocarditis. These cases suggest that HHV-6 infection should be considered as an etiology of myocarditis in immunosuppressed patients regardless of correlating blood levels. Early treatment of HHV-6 in patients with myocarditis could improve morbidity and mortality.
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Project description:Various clinical presentations of the 2019 coronavirus disease (COVID-19) have been described, including post-infectious acute and fulminant myocarditis. Here, we describe the case of a young patient admitted for COVID-19-associated post-infectious fulminant myocarditis. Despite optimal pharmacologic management, haemodynamic status worsened requiring support by veno-arterial extracorporeal membrane oxygenation. Emergent heart transplantation was required at Day 11 given the absence of cardiac function improvement. The diagnosis of post-infectious COVID-19-associated myocarditis was made from both pathologic examination of the explanted heart and positive SARS-CoV-2 serology.
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Project description:Eosinophilic myocarditis is a rare form of myocarditis characterized by eosinophilic infiltration and usually associated with peripheral hypereosinophilia. The clinical spectrum of eosinophilic myocarditis ranges widely, from mildly symptomatic to fulminant disease. When patients have fulminant eosinophilic myocarditis, high-dose corticosteroids can lead to dramatic improvement and peripheral eosinophil counts are used as an indicator of response to treatment. However, in some patients, peripheral eosinophilia is absent at initial presentation; reaching a diagnosis and determining treatment response can be challenging in this situation. This report describes a patient with fulminant eosinophilic myocarditis who initially presented with a normal peripheral eosinophil count, was diagnosed through an early endomyocardial biopsy, and was successfully treated with corticosteroids. Endomyocardial biopsy should be performed to confirm the presence of myocardial eosinophilic infiltration, especially for patients who present with fulminant myocarditis, even when peripheral eosinophilia is absent.
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Project description:BackgroundAcute fulminant myocarditis in children is associated with elevated mortality and morbidity with few advances in its medical management. Here we report a preliminary experience of children treated with IL-1 receptor antagonist associated with rapid myocardial function recovery.MethodsA retrospective case series of children admitted in the Pediatric Intensive Care Unit of the Bicêtre Hospital (AP-HP Paris Saclay University) between April 2020 and January 2022 with acute myocarditis. Children were treated with subcutaneous anakinra (an IL-1 receptor antagonist). Patients characteristics, and outcome are reported.ResultsOf 10 children admitted with acute fulminant myocarditis, eight were treated with sub-cutaneous anakinra. Seven children had SARS-CoV-2 post-infective myocarditis associated with multisystem inflammatory syndrome in children (MIS-C) and one child Parvovirus B19 myocarditis. In all patients a rapid (< 24 h) improvement in myocardial function was observed with concomitant decrease in myocardial enzymes. All patients survived with full myocardial recovery.ConclusionsIn this pilot study, use of IL-1 receptor antagonist in the initial treatment of acute fulminant myocarditis in children seems to be associated with rapid stabilization and recovery.
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Project description:We report a case of cardiac recovery from coronavirus disease 2019 (COVID-19)-associated fulminant myocarditis in a 48-year-old woman diagnosed with COVID-19 infection 4 days before, whose hemodynamic collapse were resuscitated first with venoarterial extracorporeal membranous oxygenation, followed by escalation to extracorporeal biventricular assist devices (ex-BiVAD) using two centrifugal pumps and an oxygenator. She was likely to be multisystem inflammatory syndrome in adults (MIS-A) negative. Cardiac contractility gradually recovered after the 9th day of ex-BiVAD support, and the patient was successfully weaned from ex-BiVAD on the 12th day of support. Due to postresuscitation encephalopathy, she was transferred to the referral hospital for rehabilitation with recovered cardiac function. The histopathology of the myocardial tissue showed smaller amounts of lymphocytes and more infiltration of macrophages. It is important to recognize two phenotypes of MIS-A+ or MIS-A−, with distinct manifestations and outcomes. It is also important to refer urgently such patients with COVID-19-associated fulminant myocarditis, showing different histopathology from usual viral myocarditis, with evolution toward refractory cardiogenic shock to a center with capability for advanced mechanical support to avoid a too-late cannulation. Learning objective We should recognize the clinical course and histopathology of the multisystem inflammatory syndrome in adults phenotype of coronavirus disease 2019-associated fulminant myocarditis. We should urgently refer such patients with evolution toward refractory cardiogenic shock to a center with capability for advanced mechanical support, such as venoarterial extracorporeal membrane oxygenation, Impella (Abiomed, Danvers, MA, USA), and extracorporeal biventricular assist devices.
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Project description:Immune checkpoint inhibitors have improved clinical outcomes associated with numerous cancers, but high-grade, immune-related adverse events can occur, particularly with combination immunotherapy. We report the cases of two patients with melanoma in whom fatal myocarditis developed after treatment with ipilimumab and nivolumab. In both patients, there was development of myositis with rhabdomyolysis, early progressive and refractory cardiac electrical instability, and myocarditis with a robust presence of T-cell and macrophage infiltrates. Selective clonal T-cell populations infiltrating the myocardium were identical to those present in tumors and skeletal muscle. Pharmacovigilance studies show that myocarditis occurred in 0.27% of patients treated with a combination of ipilimumab and nivolumab, which suggests that our patients were having a rare, potentially fatal, T-cell-driven drug reaction. (Funded by Vanderbilt-Ingram Cancer Center Ambassadors and others.).
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Project description:Fulminant myocarditis (FM) is an uncommon syndrome characterized by sudden and severe hemodynamic compromise secondary to acute myocardial inflammation, often presenting as profound cardiogenic shock, life-threatening ventricular arrhythmias and/or electrical storm. FM may be refractory to conventional therapies and require mechanical circulatory support (MCS). The immune system has been recognized as playing a pivotal role in the pathophysiology of myocarditis, leading to an increased focus on immunosuppressive treatment strategies. Recent data have highlighted not only the fact that FM has significantly worse outcomes than non-FM, but that prognosis and management strategies of FM are heavily dependent on histological subtype, placing greater emphasis on the role of endomyocardial biopsy in diagnosis. The impact of subtype on severity and prognosis will likewise influence how aggressively the myocarditis is managed, including whether MCS is warranted. Many patients with refractory cardiogenic shock secondary to FM end up requiring MCS, with venoarterial extracorporeal membrane oxygenation demonstrating favorable survival rates, particularly when initiated prior to the development of multiorgan failure. Among the challenges facing the field are the need to more precisely identify immunopathophysiological pathways in order to develop targeted therapies, and the need to better optimize the timing and management of MCS to minimize complications and maximize outcomes.
| S-EPMC8915421 | biostudies-literature
Project description:BackgroundFulminant myocarditis is a catastrophic disease with high mortality and complications. A viral aetiology is frequent and the implication of SARS-CoV-2 is not yet known.Case summaryA 38-year-old woman who recently arrived from Spain presented with palpitations that started suddenly 3 days prior to presentation and were associated with haemodynamic instability, without dyspnoea or chest pain. We found features of myopericarditis on the electrocardiogram and severe systolic dysfunction on the echocardiogram. The chest tomography showed findings which suggested COVID-19 infection, and PCR for SARS-CoV-2 was positive. The cardiac magnetic resonance image showed Lake Louise criteria for myocarditis. The patient was treated with immunomodulatory, steroid, and immunoglobulin therapy, with a favourable clinical response.DiscussionThe importance of this case lies in highlighting the severe cardiac involvement in a young patient, without previous risk factors, positive for COVID-19, and the favourable response to the medical treatment given.
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