Case report
Endocarditis due to Trichosporon beigelii 11 years after mitral valve replacement
Endocardite por Trichosporon beigelii 11 anos após cirurgia de troca valvar mitral
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Coutinho, Manuel J. Antunes" "autores" => array:2 [ 0 => array:2 [ "nombre" => "Gonçalo F." "apellidos" => "Coutinho" ] 1 => array:2 [ "nombre" => "Manuel J." 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AE: left atrium; AD: right atrium; PM: mitral bioprosthetic valve; VD: right ventricle; VE: left ventricle.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Trichosporon beigelii</span> is a non-pathogenic fungus that causes superficial infections such as white piedra.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Systemic infection is rare but is much more serious, with mortality approaching 80%.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Eleven cases of infective endocarditis (IE) due to <span class="elsevierStyleItalic">T. beigelii</span> have been reported.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–11</span></a> Most were in immunocompetent patients whose only risk factor was the presence of a prosthetic heart valve. Of these 11 cases, nine presented endocarditis on a prosthetic valve,<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3,5–11</span></a> in five of which the prosthetic mitral valve was affected.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,7–11</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">The longest interval between surgery and IE of a prosthetic valve reported in the literature was eight years. In the present study, a case of prosthetic valve IE due to <span class="elsevierStyleItalic">T. beigelii</span> is reported in an immunocompetent patient 11 years after valve replacement.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">A 57-year-old man with a bioprosthetic mitral valve implanted 11 years previously was admitted with a history of high fever and chills in the previous 15 days. He denied intravenous drug abuse, diabetes, malignancy or other immunosuppressive conditions. On physical examination he was febrile (axillary temperature 38.4<span class="elsevierStyleHsp" style=""></span>°C), with blood pressure of 125/78<span class="elsevierStyleHsp" style=""></span>mmHg and heart rate of 118<span class="elsevierStyleHsp" style=""></span>bpm. Cardiovascular assessment showed regular heart rhythm, normal heart sounds and a significant systolic murmur in the mitral focus. The rest of the examination was normal.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Laboratory tests showed slight leukocytosis without eosinophilia. Transthoracic echocardiography revealed an image suggesting vegetations on the biological valve, which was confirmed by transesophageal echocardiography (<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>). Blood cultures were taken and empirical antimicrobial therapy was begun, but the patient’s infection worsened and he developed hemodynamic instability. The mitral bioprosthesis was accordingly replaced by a metal valve. The vegetation adhering to the bioprosthesis was cultured and following macroscopic examination (<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>) amphotericin B was added. On the sixth postoperative day growth of <span class="elsevierStyleItalic">T. beigelii</span> was observed in the culture from the bioprosthesis and, in view of clinical deterioration, the antifungal was replaced by voriconazole. However, the patient developed multiorgan failure and died on the 13th day after surgical intervention.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">In the literature, there are only 11 published cases of IE due to <span class="elsevierStyleItalic">T. beigelii.</span><a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–11</span></a> In the nine reports of cases affecting prosthetic valves,<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3,5–11</span></a> the mean interval between cardiac surgery and the diagnosis of IE was four years, ranging between three months and eight years. The present case is unusual in that 11 years had passed after valve replacement, making this the longest period so far recorded.</p><p id="par0035" class="elsevierStylePara elsevierViewall">There are no laboratory findings characteristic of trichosporonemia,<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3</span></a> which often delays diagnosis of this serious condition. Eosinophilia was observed in only four<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,5,6,8</span></a> of the 11 reported cases and was not seen in the present case.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Blood cultures are frequently negative in systemic trichosporonosis,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> although they revealed the presence of <span class="elsevierStyleItalic">Trichosporon</span> spp. in seven of the reported cases,<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3,5,7</span></a><span class="elsevierStyleSup">,</span><a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9–11</span></a> and in those with negative blood cultures, cultures from other sites confirmed the infection.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a>,<a class="elsevierStyleCrossRef" href="#bib0020">4</a>,<a class="elsevierStyleCrossRef" href="#bib0030">6</a>,<a class="elsevierStyleCrossRef" href="#bib0040">8</a> This was also the case in our patient, in whom the organism was only isolated from a culture of the valve vegetation.</p><p id="par0045" class="elsevierStylePara elsevierViewall">The current first-line treatment of IE due to <span class="elsevierStyleItalic">Trichosporon</span> spp. is valve replacement associated with antifungal therapy.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> The antifungal used in most of the reported cases was amphotericin B,<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2–8,10,11</span></a> although some authors suggest others, including voriconazole. Analysis of the effectiveness of antifungal therapy is hindered by the severity of patients suffering from <span class="elsevierStyleItalic">T. beigelii</span> fungemia, in whom mortality can reach 80%.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Only four of the 11 reported cases were considered cured during hospital stay,<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,7,9,11</span></a> while eight patients died during four years of medical follow-up.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2–7,9,10</span></a></p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0050" class="elsevierStylePara elsevierViewall">Of the few cases of IE of prosthetic valves due to <span class="elsevierStyleItalic">T. beigelii</span>, the current case presents the longest interval between valve replacement surgery and diagnosis of infection by this pathogen.</p><p id="par0055" class="elsevierStylePara elsevierViewall">As in all the cases in the literature, low clinical suspicion led to a delay in beginning appropriate therapy. Considering the high mortality and severity of <span class="elsevierStyleItalic">T. beigelii</span> endocarditis, it should be considered when there is a prosthetic valve infection with negative blood cultures, irrespective of the time elapsed since the previous surgery.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Conflicts of interest</span><p id="par0060" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:3 [ "identificador" => "xres1497285" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1359454" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1497284" "titulo" => "Resumo" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1359455" "titulo" => "Palavras chave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Case report" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Conclusion" ] 8 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflicts of interest" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2017-11-30" "fechaAceptado" => "2018-04-08" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1359454" "palabras" => array:3 [ 0 => "Endocarditis" 1 => "Heart valve prosthesis" 2 => "Trichosporon" ] ] ] "pt" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palavras chave" "identificador" => "xpalclavsec1359455" "palabras" => array:3 [ 0 => "Endocardite" 1 => "Prótese valvar" 2 => "<span class="elsevierStyleItalic">Trichosporon</span>" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleItalic">Trichosporon beigelii</span> is a non-pathogenic fungus that can however become an opportunist agent of disseminating and potentially fatal infections, especially in immunocompromised patients. In the literature, there are only 11 published cases of infective endocarditis due to <span class="elsevierStyleItalic">T. beigelii</span>. Most of these cases involved immunocompetent individuals and the main risk factor was the presence of a prosthetic valve. The longest interval between surgery and endocarditis was eight years. In the present study, a case of prosthetic valve endocarditis due to <span class="elsevierStyleItalic">T. beigelii</span> is reported in an immunocompetent patient 11 years after mitral valve replacement. As with similar cases, low clinical suspicion and negative blood cultures delayed the beginning of antifungal therapy and cardiac surgery. Considering the high mortality and severity of <span class="elsevierStyleItalic">T. beigelii</span> endocarditis, it should be considered when there is a prosthetic valve infection with negative blood cultures, irrespective of the time elapsed since the previous surgery.</p></span>" ] "pt" => array:2 [ "titulo" => "Resumo" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleItalic">Trichosporon beigelii</span> é um fungo não patogênico, que, entretanto, pode se tornar um agente oportunista de infecções disseminadas e potencialmente fatais, especialmente em pacientes imunossuprimidos. Na literatura, foram relatados apenas onze casos de endocardite infecciosa (EI) por <span class="elsevierStyleItalic">T</span>. <span class="elsevierStyleItalic">beigelii,</span> sendo a maioria dos indivíduos imunocompetentes, cujo único fator de risco era a presença de prótese valvar. O maior intervalo registrado entre a cirurgia e a endocardite foi de oito anos. No presente estudo, será relatado um caso de endocardite de valva protética por <span class="elsevierStyleItalic">Trichosporon beigelii</span> em paciente imunocompetente, onze anos após troca valvar mitral. Como nos demais relatos, a baixa suspeição clínica e hemoculturas negativas levaram ao atraso no início da terapia antifúngica e cirurgia cardíaca. Devido à alta gravidade e mortalidade da endocardite por <span class="elsevierStyleItalic">Trichosporon beigelii</span>, este relato de caso sugere que a mesma deve ser considerada nos casos de infecção de valva protética, mesmo com hemoculturas negativas, independentemente do tempo da intervenção.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Couto R, Couto G, Abrahão I, Compagnoni I, Carnio T, Tolentino J, Endocardite por <span class="elsevierStyleItalic">Trichosporon beigelii</span> 11 anos após cirurgia de troca valvar mitral. Rev Port Cardiol. 2021;40:305.</p>" ] ] "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1169 "Ancho" => 1583 "Tamanyo" => 185284 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0070" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Transesophageal echocardiogram with images suggesting mobile vegetations on the mitral valve prosthesis (arrows). AE: left atrium; AD: right atrium; PM: mitral bioprosthetic valve; VD: right ventricle; VE: left ventricle.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1476 "Ancho" => 1476 "Tamanyo" => 291298 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0075" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Surgical specimen of the mitral valve prosthesis (left ventricular face). Arrows show friable vegetations adhering to the three valve leaflets.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:11 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "A case of <span class="elsevierStyleItalic">Trichosporon beigelii</span> endocarditis" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:3 [ 0 => "M. Mooty" 1 => "S. Kanj" 2 => "M. 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