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Sánchez Quirós, N. Ruiz López, R. López Herrero, C. Bartolomé Bartolomé" "autores" => array:4 [ 0 => array:4 [ "nombre" => "B." "apellidos" => "Sánchez Quirós" "email" => array:1 [ 0 => "Belensq93@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "N." "apellidos" => "Ruiz López" ] 2 => array:2 [ "nombre" => "R." "apellidos" => "López Herrero" ] 3 => array:2 [ "nombre" => "C." "apellidos" => "Bartolomé Bartolomé" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Anestesiología y Reanimación, Hospital Clínico Universitario de Valladolid, Valladolid, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Endocarditis marántica" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 782 "Ancho" => 800 "Tamanyo" => 49578 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Ischaemic stroke in the territory of the right carotid artery.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Nonbacterial thrombotic endocarditis (NBTE), also known as marantic endocarditis, is a rare disease characterised by the presence of sterile growths and fibrin deposits on predominantly left heart valves. It is associated with a hypercoagulable state secondary to systemic diseases such as cancer, autoimmune diseases and HIV. Its main manifestations are stroke, pulmonary thromboembolism, acute intestinal ischaemia, and splenic, renal, and hepatic infarction. Despite this, it is frequently under-diagnosed, and most cases are discovered post-mortem.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">A 57-year-old woman with a history of untreated dyslipidaemia and uterine neoplasia who underwent surgery 5 years previously, presented with left hemiplegia and oculocephalic deviation to the right. She had had a flu-like illness the previous week with fever of up to 38<span class="elsevierStyleHsp" style=""></span>°C. Axial computed tomography (CT) showed loss of corticosubcortical differentiation affecting the right cerebral hemisphere, suggesting an established iscahemic lesion compatible with acute ischaemic stroke of unknown origin in the right carotid territory (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The patient was considered a candidate for endovascular reperfusion therapy and thrombectomy was performed.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Four days later she was admitted to the intensive care unit for an episode of acute respiratory failure, which was resolved with non-invasive mechanical ventilation for 6<span class="elsevierStyleHsp" style=""></span>h. Antio-CT showed filling defects in both lower lobe arteries, consistent with acute pulmonary thromboembolism. Six hours later she debuted with paroxysmal atrial fibrillation (AF) with rapid ventricular response; she was hypertensive from the start and responded well to labetalol. She also presented Osler's nodes and Janeway lesions, predominantly on the lower extremities (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). A transthoracic echocardiogram (TTE) was performed, showing severe mitral regurgitation. Transoesophageal echocardiography (TOE) confirmed the diagnosis of endocarditis with grade 2–3 mitral regurgitation and threadlike structure arising from the mitral valve. Suspecting endocarditis, blood cultures were taken and empirical antibiotic treatment with vancomycin and gentamicin was started, to which ceftriaxone was added until pulmonary infection could be ruled out. Bronchoalveolar lavage, blood cultures, and urine cultures were negative. Samples were drawn to determine the presence of antiphospholipid, anti-DNA, C3 and C4 antibodies, which were negative.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Given the increase in tumour markers (CEA: 550<span class="elsevierStyleHsp" style=""></span>ng/ml, CA 19.9: 34.335<span class="elsevierStyleHsp" style=""></span>IU/ml, CA 125: 28.2<span class="elsevierStyleHsp" style=""></span>IU/ml) a full body CT scan was requested (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>), which showed an enlarged liver secondary to multiple hypodense lesions distributed throughout the parenchyma, at first suggesting metastatic spread. We also observed areas of decreased uptake in the renal cortex of both kidneys and the splenic parenchyma, compatible with infarction. The patient was transferred to the Cardiology ward with stable haemodynamic and respiratory variables and a diagnosis of marantic endocarditis.</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">A week later, she presented abdominal pain and bloating suggestive of intestinal ischaemia and underwent surgery, which confirmed the suspicion. Given the presence of massive small bowel ischaemia, no resection was performed and the patient was admitted to the Critical Care Unit, where heparin sodium infusion was started. Tumour markers had increased, reaching CEA: 1080<span class="elsevierStyleHsp" style=""></span>ng/ml, CA 19.9: >50,000<span class="elsevierStyleHsp" style=""></span>IU/ml and CA 125: 150<span class="elsevierStyleHsp" style=""></span>IU/ml. The patient did not respond to treatment, and her condition rapidly deteriorated; she died 72<span class="elsevierStyleHsp" style=""></span>h later due to multiorgan failure.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">NBTE or marantic endocarditis is caused by a hypercoagulable state secondary to a series of prothrombotic diseases such as cancer (mainly of the pancreas, lung, and ovary), autoimmune diseases (lupus erythematosus, antiphospholipid syndrome), or HIV infection.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">1</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">It is thought to originate from the interaction between monocytes or macrophages and malignant cells, which results in the release of tumour necrosis factor and interleukins, triggering endothelial damage and the formation of a thrombogenic vascular surface.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">2</span></a> Increased expression of tissue factor on monocytes and endothelial cells causes activation of the extrinsic pathway and increases thrombin production. This causes an imbalance in the prothrombotic and fibrinolytic mechanisms similar to that which occurs in disseminated intravascular coagulation, and promotes the growth of small sterile fibrin vegetations on the heart valve that cause a series of systemic embolic events.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Cerebral strokes are the most frequent presentation, and in many cases are the debut symptom of the underlying disease. A quarter of all strokes in cancer patients are caused by NBTE.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">4</span></a> Neurological events are the main cause of death in these patients,<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">5</span></a> although NBTE is also associated with other pathologies, such as acute pulmonary thromboembolism, intestinal ischaemia, splenic and renal infarcts, and peripheral embolisms, such as Osler's nodes and Janeway lesions. Histologic evidence of disseminated intravascular coagulation has been found in 50% of patients with NBTE, together with hyperfibrinogenaemia, thrombocytopaenia, high levels of fibrin degradation products and low levels of antithrombin III.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">6</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">TOE is the diagnostic procedure of choice in this type of patient because NBTE vegetations are typically small and TTE is inconclusive in most cases, including our patient, due to the low sensitivity that this technique for vegetations measuring less than 5 mm.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">7</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">It is important to perform differential diagnosis with infective endocarditis and Libman–Sacks endocarditis that is typical of lupus and also presents as sterile vegetation with no evidence of infection.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">7</span></a> The use of the modified Duke criteria, tests for antiphospholipid antibodies and echocardiogram findings can help distinguish between these entities.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">8</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Treatment should target the underlying malignancy, and anticoagulants must be administered to avoid recurrent systemic embolisms. Surgery is only indicated in patients with thromboembolic events despite anticoagulant therapy, or those with severe valvular dysfunction that can lead to heart failure. Early anticoagulation with sodium heparin or low molecular weight heparin (LMWH) appears to reduce the risk of mortality in these patients.<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">9</span></a> LMWH appears to be more effective than oral anticoagulants. Despite this, the outcome is early mortality in most patients, particularly those with advanced stage tumours.</p><p id="par0060" class="elsevierStylePara elsevierViewall">In our case, the patient debuted with an ischaemic stroke, the most frequent clinical presentation NBTE, as mentioned above. This was followed by other ischaemic events, including pulmonary thromboembolism, Janeway lesions, and Osler's nodes. After discussing the case with the multidisciplinary team from the Stroke unit we considered starting systemic anticoagulants. However, given the high risk of haemorrhagic transformation shortly after ischaemic stroke, and the fact that respiratory failure resolved within a few hours, we decided against this strategy.</p><p id="par0065" class="elsevierStylePara elsevierViewall">After the onset of atrial fibrillation and arterial hypertension, and given the suspicion of endocarditis, we decided to administered beta-blockers to control heart rate. Amiodarone was ruled out due to the risk of conversion to sinus rhythm and the possible presence of thrombi in the atrium.<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">10</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">TTE showed mitral regurgitation, although the vegetation in the mitral valve was only visible on TOE due to its small size, typical of NBTE. Once endocarditis had been diagnosed, empirical antibiotic therapy for native valve endocarditis was started (vancomycin<span class="elsevierStyleHsp" style=""></span>+<span class="elsevierStyleHsp" style=""></span>gentamicin).<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">11</span></a> Due to the patient's history of respiratory infection in the days prior to admission, ceftriaxone was added to this due while awaiting microbiological results. When these were received, the diagnosis of noninfectious endocarditis or NBTE was established. CT showed liver lesions suggestive of metastasis, and the increase in tumour markers indicated a recurrence of the previous uterine tumour as the origin of the condition.</p><p id="par0075" class="elsevierStylePara elsevierViewall">The diagnosis of intestinal ischaemia was made 10 days after the stroke, and we decided to take a conservative approach. The risks associated with heparin administration had reduced considerable once the risk of haemorrhagic transformation of stroke had passed, and was a good therapeutic option.<a class="elsevierStyleCrossRef" href="#bib0120"><span class="elsevierStyleSup">12</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">NBTE is a frequently underdiagnosed pathology that must be considered in cancer patients who debut with thromboembolic phenomena. Early diagnosis and treatment are essential in order to reduce the risk of NBTE-related comorbidities and improve survival.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:8 [ 0 => array:3 [ "identificador" => "xres1330894" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1226241" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1330895" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1226240" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Case report" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2019-05-08" "fechaAceptado" => "2020-01-14" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1226241" "palabras" => array:6 [ 0 => "Marantic endocarditis" 1 => "Nonbacterial thrombotic endocarditis" 2 => "Anticoagulation" 3 => "Diagnosis" 4 => "Treatment" 5 => "Transesophageal echocardiography" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1226240" "palabras" => array:7 [ 0 => "Endocarditis marántica" 1 => "Endocarditis trombótica no bacteriana" 2 => "Anticoagulación" 3 => "Diagnóstico" 4 => "Tratamiento" 5 => "Ecocardiograma transesofágico" 6 => "Endocarditis tumoral" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Marantic endocarditis is characterised by the presence of sterile vegetations in the heart valves, and is associated with hypercoagulability states (cancer, autoimmune diseases, HIV). Its main complications are stroke, pulmonary thromboembolism, acute intestinal ischaemia and splenic, renal and hepatic infarcts. We present the case of a 57-year-old patient with a history of uterine neoplasia. She went to the emergency department due to sudden loss of strength in the left side of the body. A computed tomography (CT) scan showed right ischaemic stroke, and she underwent endovascular reperfusion and thrombectomy. Four days later, she suffered acute respiratory failure, with angio-CT showing pulmonary thromboembolism. Later, paroxysmal atrial fibrillation and distal ischaemia in the second toe of the left foot appeared. She was diagnosed with marantic endocarditis by means of transesophageal echocardiography, and died 72<span class="elsevierStyleHsp" style=""></span>h later due to multiorgan failure. Early diagnosis and treatment with anticoagulation can reduce the mortality of this disease, since it is underdiagnosed, and often only comes to light during postmortem examination.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">La endocarditis marántica se caracteriza por la presencia de vegetaciones estériles en las válvulas cardiacas. Se asocia a estados de hipercoagulabilidad (cáncer, enfermedades autoinmunes, VIH). El ictus, el tromboembolismo pulmonar, la isquemia intestinal aguda y los infartos esplénicos, renales y hepáticos son sus principales manifestaciones. Presentamos el caso de una paciente de 57 años, con antecedentes de neoplasia uterina intervenida 5 años antes, que acudió al servicio de Urgencias por pérdida brusca de fuerza en hemicuerpo izquierdo. La tomografía axial computarizada mostró un ictus isquémico derecho y se sometió a reperfusión endovascular y trombectomía 3<span class="elsevierStyleHsp" style=""></span>h después del inicio de la clínica. A los 4 días sufrió insuficiencia respiratoria aguda, con angio-TAC compatible con tromboembolismo pulmonar. Después aparecieron fibrilación auricular paroxística e isquemia distal en el segundo dedo del pie izquierdo. Fue diagnosticada de endocarditis marántica por ecocardiograma transesofágico y falleció 72<span class="elsevierStyleHsp" style=""></span>h después por fracaso multiorgánico. El diagnóstico y el tratamiento precoces con anticoagulación pueden disminuir la mortalidad de esta enfermedad, que suele ser infradiagnosticada, con un alto porcentaje de diagnóstico post mortem.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Sánchez Quirós B, Ruiz López N, López Herrero R, Bartolomé Bartolomé C. Endocarditis marántica. Rev Esp Anestesiol Reanim. 2020;67:208–211.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 782 "Ancho" => 800 "Tamanyo" => 49578 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Ischaemic stroke in the territory of the right carotid artery.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 431 "Ancho" => 1500 "Tamanyo" => 65229 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Janeway lesions and Osler's nodes on the right hand and left foot.</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 626 "Ancho" => 1500 "Tamanyo" => 88753 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Hypodense lesions in the liver parenchyma compatible with liver metastases.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:12 [ 0 => array:3 [ "identificador" => "bib0065" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Non-infective endocarditis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "A. 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Case report
Marantic endocarditis
Endocarditis marántica
B. Sánchez Quirós
, N. Ruiz López, R. López Herrero, C. Bartolomé Bartolomé
Corresponding author
Servicio de Anestesiología y Reanimación, Hospital Clínico Universitario de Valladolid, Valladolid, Spain