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Case Report
Papillary fibroelastoma of aortic valve mimicking an infective endocarditis
Fibroelastoma papilar de válvula aórtica que simula una endocarditis infecciosa
Reda Bzikhaa,b,
Corresponding author
redabzikha@gmail.com

Corresponding author.
, Amin Serradja,b, Sébastien Querona,b
a Université Paris Descartes, Sorbonne Paris Cité, Paris 75006, France
b Department of Cardiovascular Surgery, AP-HP, European Georges Pompidou Hospital, Paris 75015, France
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Papillary fibroelastomas are benign tumors that can develop on any endocardial surface&#46;<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">1&#44;2</span></a> However&#44; they are usually found on the cardiac valves&#44; and most frequently on the aortic valve&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">3</span></a> Even though these tumors are categorized as benign and are usually asymptomatic&#44; those in the right side of the heart can cause pulmonary embolism&#44; while those in the left side can cause serious complications&#46;<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">4&#44;5</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">We report a case of a 76-year-old woman who had a permanent pacemaker since October 2018 for sinus node dysfunction&#44; and who presented at a regional hospital with a one-week history of fever associated with palpitations&#46; Her first transthoracic echocardiography showed an image of suspicious vegetation&#46; And during her hospitalization she received a one-week course of antibiotics to treat a culture-negative endocarditis&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Then&#44; she was transferred to our hospital for further investigations&#46; In our department&#44; the transesophageal echocardiography revealed a mobile&#44; hypoechoic&#44; heterogeneous mass&#44; oval shaped&#44; appended on the aortic surface of the right coronary cusp&#44; measuring 14<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>8<span class="elsevierStyleHsp" style=""></span>mm&#46; This mass was compatible with both papillary fibroelastoma and vegetation &#40;<a class="elsevierStyleCrossRefs" href="#fig0005">Figs&#46; 1&#8211;3</a>&#41;&#46; Fortunately&#44; no other mass was found on the leads of the pacemaker&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Based on medical history&#44; clinical exam&#44; echocardiographic image&#44; negative blood cultures&#44; and laboratory tests findings&#44; the most likely diagnosis was a culture-negative endocarditis&#44; a fibroelastoma of the aortic valve&#44; or both&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">In order to reduce the risk of embolism as well as to confirm an etiological diagnosis we scheduled an emergency surgical removal of the mass&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">We initiated cardiopulmonary bypass by cannulating the distal ascending aorta and the right atrium with a dual stage venous cannula&#46; After cross-clamping of the aorta&#44; we injected a retrograde cold crystalloid cardioplegia&#44; then we made a transverse aortotomy to expose the mass&#44; which was soft&#44; measuring 10<span class="elsevierStyleHsp" style=""></span>mm and adhering to the free edge of the right coronary cusp of the aortic valve&#46; We excised it carefully from the surface of the leaflet&#44; ensuring that no mass was left behind&#46; Then&#44; we inspected the ascending aorta and the left ventricle for other tumors or vegetations&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">The absence of aortic valve dysfunction was confirmed intraoperatively before the aortotomy was closed&#46; After deairing maneuvers&#44; we released the aortic cross-clamp&#46; The patient&#39;s body was rewarmed&#44; spontaneous cardiac contractility resumed with sinus pause as it was preoperatively&#44; requiring the reactivation of the pacemaker&#44; then the patient was successfully weaned from cardiopulmonary bypass&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">The intraoperative transesophageal echocardiography showed the absence of any residual mass&#44; without any leakage of the aortic valve&#46; Histological and microbiological investigations confirmed the nature of the papillary fibroelastoma of the mass&#44; with no evidence of infective endocarditis &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46;</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0045" class="elsevierStylePara elsevierViewall">The follow-up was uneventful&#44; and no residual or recurrent tumor had been seen in the follow-up transthoracic echocardiography&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0050" class="elsevierStylePara elsevierViewall">Papillary fibroelastomas are small benign tumors that represent 10&#37; of all cardiac tumors&#44;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">1</span></a> and measure 9&#8211;12<span class="elsevierStyleHsp" style=""></span>mm&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">2</span></a> Histologically&#44; their constituting layers have a similarity to those of the chordae tendineae&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">6</span></a> They can develop on any endocardial surface&#46;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">7</span></a> 90&#37; of the cases are attached to the aortic valve&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">3</span></a> Even though they are categorized as benign tumors and in the majority of cases they are asymptomatic&#46; Those in the right-side of the heart can cause pulmonary embolism&#44; while those in the left-side can cause serious complications&#44; such as systemic embolic events&#44; stroke&#44; and acute myocardial infarction&#46;<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">4&#44;5</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The mechanism of these complications could be either by the fragmentation of the papillary fibroelastoma&#44; or by the disintegration of the thrombi that have adhered to the surface of these tumors&#46; Also&#44; there is another mechanism that is the prolapse of the tumor itself into any coronary ostium leading to an acute myocardial infarction&#46;<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">4&#44;5&#44;8</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">The transesophageal echocardiography is superior to the transthoracic one in the detection&#44; the localization and the description of the cardiac masses and it has better visualization of valves and cardiac chambers&#46;<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">9</span></a> Typically&#44; the papillary fibroelastoma is seen as<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">2</span></a>&#58;<ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">-</span><p id="par0065" class="elsevierStylePara elsevierViewall">Small round or oval tumor of 9&#8211;12<span class="elsevierStyleHsp" style=""></span>mm in diameter&#46;</p></li><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">-</span><p id="par0070" class="elsevierStylePara elsevierViewall">Homogeneous speckled texture&#46;</p></li><li class="elsevierStyleListItem" id="lsti0015"><span class="elsevierStyleLabel">-</span><p id="par0075" class="elsevierStylePara elsevierViewall">Stippling along its edges&#46;</p></li><li class="elsevierStyleListItem" id="lsti0020"><span class="elsevierStyleLabel">-</span><p id="par0080" class="elsevierStylePara elsevierViewall">Mobile if it has a pedicle&#46;</p></li><li class="elsevierStyleListItem" id="lsti0025"><span class="elsevierStyleLabel">-</span><p id="par0085" class="elsevierStylePara elsevierViewall">And 50&#37; of them have small stalks&#46;</p></li></ul></p><p id="par0090" class="elsevierStylePara elsevierViewall">Papillary fibroelastomas can be confused with other cardiac tumors&#44; vegetations&#44; thrombi&#44; valvular calcifications&#44; Libman&#8211;Sacks and Lambl&#39;s excrescences&#44; leading to an incorrect therapeutic management&#46;</p><p id="par0095" class="elsevierStylePara elsevierViewall">Concerning cardiac tumors&#44; there are several ones that can mimic papillary fibroelastomas&#44; such as myxoma that is a multi-lobed one and that is located on the cardiac walls&#44; mostly in the left atrium&#44; and in the interatrial septum&#46; Histologically&#44; it has polygonal myxoma cells and multiple blood vessels within the papillae&#46;<a class="elsevierStyleCrossRef" href="#bib0120"><span class="elsevierStyleSup">10</span></a></p><p id="par0100" class="elsevierStylePara elsevierViewall">The atrial thrombi are usually located in the atrial appendage&#44; and mostly associated with atrial fibrillation&#44; and atrial dilatation&#46; However&#44; the ventricular ones are typically located in the ventricular aneurysm&#44; in the akinesis wall of the infarcted myocardium&#44; and in any cardiac wall in case of the cardiomyopathy&#46; In this case the echocardiography shows an irregular border&#44; and the absence of a pedicle&#46;</p><p id="par0105" class="elsevierStylePara elsevierViewall">As in our case the principal differential diagnosis of papillary fibroelastoma was a vegetation&#44; which made the therapeutic management challenging&#46; It is known that vegetations are usually located in cardiac valves&#44; and they are usually associated with valvular destruction&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">5</span></a> Also&#44; there is a usual association between clinical signs of endocarditis and valvular destruction&#46; Thus&#44; the echocardiography may show a mass that changes in appearance over time and may reveal signs of valvular regurgitation&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">11</span></a> We should note that the fever identified in our patient during the preoperative period remained without any explanation&#44; regardless of the tests that were done&#44; however the association between the endocarditis and the papillary fibroelastoma was not totally excluded&#46;</p><p id="par0110" class="elsevierStylePara elsevierViewall">Surgical excision is the sole curative treatment of papillary fibroelastoma&#44;<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">12</span></a> which was performed for the first time by Lichtenstein in 1979&#46;<a class="elsevierStyleCrossRef" href="#bib0135"><span class="elsevierStyleSup">13</span></a> During the procedure all cardiac chambers and valves must be inspected to eliminate other localizations of the tumor&#44; and sometimes to look for the association of a papillary fibroelastoma with vegetations&#46; Furthermore&#44; valvular regurgitation can be caused either by the tumor itself or by the surgeon after the excision&#46; In this matter&#44; the surgical procedure must be completed either by valvular repair&#44; every time the valve is repairable&#44; otherwise valvular replacement can be done&#46;<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">14</span></a></p><p id="par0115" class="elsevierStylePara elsevierViewall">For symptomatic patients&#44; surgery is highly recommended&#44; whereas for asymptomatic patients&#44; surgery should be done only if the papillary fibroelastoma is mobile&#46; If not&#44; a clinical and an echocardiographic follow-up for the patient is mandatory until the symptoms develop or the tumor becomes mobile&#46; However&#44; none of these recommendations are based on a randomized controlled study&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">2&#44;14</span></a></p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0120" class="elsevierStylePara elsevierViewall">Papillary fibroelastoma of cardiac valves can mimic vegetation making it difficult to differentiate the two&#46; Surgical removal of the mass is the only curative treatment&#46; It reduces the risk of tumoral complications and confirms the etiological diagnosis&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Ethics approval</span><p id="par0125" class="elsevierStylePara elsevierViewall">IRB and clinical trial registration approval are not required for case reports&#46;</p><p id="par0130" class="elsevierStylePara elsevierViewall">Constant of the use of medical data is obtained verbally and in written form from the patient&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Funding</span><p id="par0135" class="elsevierStylePara elsevierViewall">The authors report no involvement in the research by the sponsor that could have influenced the outcome of this work&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Conflicts of interest</span><p id="par0140" class="elsevierStylePara elsevierViewall">The authors certify that there is no conflict of interest with any financial organization regarding the material discussed in the manuscript&#46;</p></span></span>"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Papillary fibroelastomas are benign tumours&#44; usually found on cardiac valves&#46; They are attached to the aortic valve in 90&#37; of cases&#46; Even though benign and mostly asymptomatic&#44; right-sided tumours can cause pulmonary embolism&#44; while left-sided tumours can cause serious complications&#46; We report a 76-year-old woman treated initially for a suspicious infective endocarditis of the aortic valve&#44; in whom papillary fibroelastoma was histologically confirmed&#44; with no evidence of infective endocarditis&#46;</p></span>"
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      "es" => array:2 [
        "titulo" => "Resumen"
        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Los fibroelastomas papilares son tumores benignos que&#44; generalmente&#44; se encuentran en las v&#225;lvulas cardiacas&#46; El 90&#37; de ellos est&#225;n unidos a la v&#225;lvula a&#243;rtica y&#44; aunque estos sean tumores benignos&#44; la mayor&#237;a pueden ser asintom&#225;ticos&#46; Los del lado derecho pueden causar embolia pulmonar&#44; mientras que los del lado izquierdo pueden ocasionar complicaciones graves&#46; Presentamos el caso de una mujer de 76 a&#241;os tratada por sospechas de endocarditis infecciosa de la v&#225;lvula a&#243;rtica&#44; a quien se le confirm&#243; patol&#243;gicamente el fibroelastoma papilar&#44; sin evidencia de endocarditis infecciosa&#46;</p></span>"
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Transoesophageal echocardiogram of 14<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>8<span class="elsevierStyleHsp" style=""></span>mm mass attached to the free edge of a right coronary cusp&#46;</p>"
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          "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Transesophageal echocardiography reveals a 14<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>8<span class="elsevierStyleHsp" style=""></span>mm mass on the aortic valve&#46;</p>"
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          "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">3D echocardiography of the mass attached to the aortic valve&#46;</p>"
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          "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Specimen of excised mass reveals papillary fibroelastoma of the endocardium&#46;</p>"
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Article information
ISSN: 11340096
Original language: English
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es en pt

¿Es usted profesional sanitario apto para prescribir o dispensar medicamentos?

Are you a health professional able to prescribe or dispense drugs?

Você é um profissional de saúde habilitado a prescrever ou dispensar medicamentos