Clinical note
18F-FDG PET/CT in a patient with atypical presentation of cardiac angiosarcoma
PET/TC con 18F-FDG en paciente con presentación atípica de angiosarcoma cardiaco
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Proyección de máxima intensidad que muestra 3 depósitos patológicos de FDG en la región cardíaca. Uno alrededor del cayado aórtico en el receso pericárdico aórtico superior, el segundo a nivel subhiliar derecho en el receso pericárdico venoso pulmonar derecho, y el último adyacente a la cara inferior de la aurícula derecha.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "J. Mestres-Marti, E. Llinares-Tello, A. Sabaté-Llobera, L. Gràcia-Sánchez, J. Robles-Barba, C. Gámez-Cenzano" "autores" => array:6 [ 0 => array:2 [ "nombre" => "J." "apellidos" => "Mestres-Marti" ] 1 => array:2 [ "nombre" => "E." "apellidos" => "Llinares-Tello" ] 2 => array:2 [ "nombre" => "A." "apellidos" => "Sabaté-Llobera" ] 3 => array:2 [ "nombre" => "L." "apellidos" => "Gràcia-Sánchez" ] 4 => array:2 [ "nombre" => "J." "apellidos" => "Robles-Barba" ] 5 => array:2 [ "nombre" => "C." 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"apellidos" => "Goñi Gironés" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S2253654X18302166" "doi" => "10.1016/j.remn.2018.10.007" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2253654X18302166?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2253808919300096?idApp=UINPBA00004N" "url" => "/22538089/0000003800000003/v1_201905020850/S2253808919300096/v1_201905020850/en/main.assets" ] "en" => array:20 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Clinical note</span>" "titulo" => "<span class="elsevierStyleSup">18</span>F-FDG PET/CT in a patient with atypical presentation of cardiac angiosarcoma" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "183" "paginaFinal" => "185" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "J. Mestres-Marti, E. Llinares-Tello, A. Sabaté-Llobera, L. Gràcia-Sánchez, J. Robles-Barba, C. Gámez-Cenzano" "autores" => array:6 [ 0 => array:4 [ "nombre" => "J." "apellidos" => "Mestres-Marti" "email" => array:1 [ 0 => "judit.mestres@idi.gencat.cat" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "E." "apellidos" => "Llinares-Tello" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "A." "apellidos" => "Sabaté-Llobera" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "L." "apellidos" => "Gràcia-Sánchez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 4 => array:3 [ "nombre" => "J." "apellidos" => "Robles-Barba" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 5 => array:3 [ "nombre" => "C." "apellidos" => "Gámez-Cenzano" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Radiodiagnóstico-IDI Bellvitge, Hospital Universitario de Bellvitge, Hospitalet de Llobregat, Barcelona, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Medicina Nuclear-PET, IDI Bellvitge, Hospital Universitario de Bellvitge, Hospitalet de Llobregat, Barcelona, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "PET/TC con <span class="elsevierStyleSup">18</span>F-FDG en paciente con presentación atípica de angiosarcoma cardiaco" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 671 "Ancho" => 900 "Tamanyo" => 46930 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleSup">18</span>F-FDG PET/TC. Axial fusion PET/CT image. Pathological FDG deposit in the right subhilum corresponding to the right pericardial pulmonary venous recess.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Cardiac tumors are infrequent, with a prevalence of 0.02–0.056%.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a> Indeed, a cardiac mass is usually related to metastasis. Among primary cardiac tumors the most frequent are benign (i.e.: mixomas, rabomyomas, etc.), and among malignant tumors, which are extremely rare, angiosarcoma is the most common.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Likewise, primary tumors of the pericardium are rare, with a frequency of 6.7–12.8% of all tumors appearing in the heart and with a overall prevalence of 0.001–0.007% (based on case series). Similarly, metastatic pericaridal involvement or direct invasion of a cardiac tumor is much more frequent. The most frequent benign mass of the pericardium is a cyst followed by lipoma, and a malignant tumor would be a mesothelioma. Other primary malignant neoplasms include different types of sarcomas or lymphoma.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">From an anatomical point of view, the pericardium is made up of 2 layers, the external fibrous layer and the internal serous layer. The latter layer is divided into 2 layers, the visceral and parietal, delimiting the pericaridal cavity which, in normal conditions, contains 15–50<span class="elsevierStyleHsp" style=""></span>ml of fluid. In turn, there are sinuses and recesses between the large vessels which may contain fluid, and this is useful to know in order not to confound these areas with mediastinic or pulmonary disease.</p><p id="par0020" class="elsevierStylePara elsevierViewall">As in most malignant diseases early accurate diagnosis of these tumors is essential for determining patient prognosis. The first imaging tests that should be performed on suspicion of cardiothoracic disease are chest radiography and echocardiography which have a limited value in the diagnosis of cardiac tumors and require other high resolution imaging tests such as transthoracic cardiac ultrasonography, thoracic computerized tomography (CT) and cardiac magnetic resonance (MR) for correct characterization. Positron emission tomography/CT (PET/CT) has a role in the initial staging and follow-up of these patients.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical case</span><p id="par0025" class="elsevierStylePara elsevierViewall">We present the case of a 74-year-old patient with a history of hepatic cirrhosis who presented clinical manifestations of 5 months of evolution of anorexia, syncopes and progressive dyspnea, which in the last days occurred with minimum effort and associated with centrothoracic pain. Cardiac echography showed abundant anterior, inferior and lateral pericardial effusion with a maximum thickness of 35–40<span class="elsevierStyleHsp" style=""></span>mm, albeit without signs of tamponade. Pericardiocentesis was performed followed by the placement of a drain with an intitial drainage of 950<span class="elsevierStyleHsp" style=""></span>ml. Fluid with hematic-like characteristics was obtained showing a negative bacteriological study and positive cytology for malignant cells. In view of these findings a PET/CT study with <span class="elsevierStyleSup">18</span>F-fluorodeoxyglucose (<span class="elsevierStyleSup">18</span>F-FDG) was carried out in search of the primary neoplasm. Only 3 areas of pathological FDG uptake in the cardiac region were of note in the whole body scan: one around the aortic arch, the second at the right subhilum level and the last adjacent to the inferior face of the right atrium (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1–3</a>). These results established a differential diagnosis between a primary pulmonary neoplasm with lymph node involvement and a primary cardiac neoplasm. The bronchoscopy was negative directing the case to a primary cardiac neoplasm of pericardial localization. Two of the radiotracer deposits were localized in pericardial recesses; the perioaortic uptake corresponded to the superior aortic recess and the right subhilum to the right pulmonary venous recess. Finally, immunohistochemical analysis of the pericardial fluid confirmed the presence of angiosarcoma. The patient accidentally removed the pericardial drainage and within a few days was readmitted for a new episode of pericardial effusion together with ascites decompensation due to liver disease. Considering the poor general status of the patient, oncospecific treatment was ruled out and the patient evolved unfavorably and died due to cardiorespiratory arrest at one month after diagnosis.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">Histologically, angiosarcoma consists of a proliferation of endothelial cells which delimit irregular vascular spaces and anastomose between each other to make up tumor masses which invade the myocardium and the pericardium presenting areas of necrosis and hemorrhage within. This tumor mainly affects middle-aged males and may be localized in any cardiac chamber or the pericardium. It is found in the right atrium in more than 80% of the cases. Two forms of presentation have been described. The first and most common is of an irregular or nodular intracavitary mass and the second is less frequent and infiltrates the whole pericardium.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">This type of tumor normally begins with very unspecific clinical manifestations. Right cardiac failure and cardiac tamponade are the two most common scenarios as the right side of the heart is most frequently affected.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">2–4</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Cardiac angiosarcoma is a very aggressive tumor with a poor prognosis since most of these tumors are unresectable at diagnosis, and 66–89% present metastasis at onset, mainly in the mediastinal lymph nodes, pulmonary parenchyma and bone.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">2,3</span></a> Likewise, these tumors show poor response to radiotherapy and chemotherapy, with patients having a mean survival of 6–11 months, thereby making early diagnosis and treatment essential.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">1,3,5,6</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The first imaging tests usually performed in these patients is chest radiography and cardiac echography. Radiography may show the presence of cardiomegaly, alteration of the mediastinal contour, mediastinal mass, pleural effusion or tumor dissemination in the pulmonary parenchyma which is very unspecific.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a> Echocardiography can often detect pericardial effusion. Pericardiocentesis usually obtains hemorrhagic fluid and the cytological results are negative for malignant cells.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">1,3</span></a> Transthoracic ultrasonography, CT and the great anatomical definition, tissue characterization and functional evalution of mainly magnetic resonance (MR) imaging are useful for achieving the diagnosis as well as to determine tumor size, localization and its relationship with adjacent structures (valves, pericardium, among others).<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a> Finally, the definitive diagnosis is obtained by histological study.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Around 15 cases of PET/CT of primary cardiac angiosarcoma have been described in the English literature and the utility of this imaging technique in achieving the diagnosis has been suggested. The studies by Dhull et al.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a> and Rahbar et al.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a> propose a SUV<span class="elsevierStyleInf">max</span> cut-off of 3.5 to noninvasively determine the malignant nature of a cardiac tumor with a sensitivity of 100% and a specificity of 86%, while morphological studies based on radiologic techniques (MR and CT) have a sensitivity of 82% and a specifity of 86% for differentiation.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a> In our study the SUV<span class="elsevierStyleInf">max</span> of the 3 FDG deposits was also greater than this cut-off suggesting malignancy. Likewise, the utility of PET/CT has been reported for achieving correct initial staging and evaluating the extension of the primary tumoral lesion as well as the presence of distant metastasis and for follow-up after treatment.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">1,4,5</span></a> In the present case, PET/CT was useful for confirming the presence of a malignant tumoral process localized in the pericardium with multifocal involvement including 2 of the pericardial recesses and with no evidence of distant metastasis.</p><p id="par0055" class="elsevierStylePara elsevierViewall">The case presented here is of interest due to the low prevalence of primary malignant cardiac tumors and because of the atypical presentation as primary tumoral involvement of the pericardium. It is also of note to highlight the importance of having knowledge of pericardial anatomy and the different recesses around the large vessels.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:8 [ 0 => array:3 [ "identificador" => "xres1185608" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1105411" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1185609" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1105410" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Clinical case" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2018-05-06" "fechaAceptado" => "2018-08-30" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1105411" "palabras" => array:4 [ 0 => "Heart tumors" 1 => "Pericardial tumors" 2 => "Angiosarcoma" 3 => "Positron emission tomography" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1105410" "palabras" => array:4 [ 0 => "Tumores cardiacos" 1 => "Tumores pericárdicos" 2 => "Angiosarcoma" 3 => "Tomografía por emisión de positrones" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Primary cardiac or pericardial tumors are infrequent, metastatic involvement being more common. Cardiac angiosarcoma is a rare primary malignant tumor of mesenchymal origin. It entails a poor prognosis mostly due to frequent metastases at the time of diagnosis, as well as low response to onco-specific treatments.</p><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">We describe a case of a 74-year-old patient with a primary cardiac angiosarcoma with an infrequent location at pericardium level. We review the literature and the utility of <span class="elsevierStyleSup">18</span>F-FDG PET/CT in the initial staging.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Los tumores cardíacos o pericárdicos primarios son infrecuentes siendo más habitual la afectación metastásica. El angiosarcoma cardíaco es un tumor primario infrecuente de origen mesenquimal y de mal pronóstico por presentar metástasis en el momento del diagnóstico, y por su pobre respuesta a los tratamientos oncoespecíficos.</p><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Se describe el caso de una paciente de 74 años, que presenta un angiosarcoma cardíaco primario, con una localización infrecuente a nivel de pericardio. Se revisa la literatura y la utilidad de la PET/TC con <span class="elsevierStyleSup">18</span>F-FDG en su estadificación inicial.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Mestres-Marti J, Llinares-Tello E, Sabaté-Llobera A, Gràcia-Sánchez L, Robles-Barba J, Gámez-Cenzano C. PET/TC con <span class="elsevierStyleSup">18</span>F-FDG en paciente con presentación atípica de angiosarcoma cardiaco. Rev Esp Med Nucl Imagen Mol. 2019;38:183–185.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1120 "Ancho" => 900 "Tamanyo" => 50539 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleSup">18</span>F-FDG PET/CT. Maximum intensity projection showing 3 pathological FDG deposits in the cardiac region: one around the aortic arch in the superior aortic pericardial recess, the second at the right subhilum level to the right pulmonary venous recess and the last adjacent to the inferior face of the right atrium.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 594 "Ancho" => 900 "Tamanyo" => 42770 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleSup">18</span>F-FDG PET/CT. Axial fusion PET/CT image. Pathological FDG deposit around the aortic arch corresponding to the superior pericardial aortic recess.</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 671 "Ancho" => 900 "Tamanyo" => 46930 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleSup">18</span>F-FDG PET/TC. Axial fusion PET/CT image. Pathological FDG deposit in the right subhilum corresponding to the right pericardial pulmonary venous recess.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0040" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Primary pericardial tumors" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "C.S. Restrepo" 1 => "D. Vargas" 2 => "D. Ocazionez" 3 => "S. Martínez-Jiménez" 4 => "S.L. Betancourt Cuellar" 5 => "F.R. 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