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CASE REPORT
Unusual presentation of multiple aneurysms of the ascending aorta
Sergio Francisco Santos Jr.I,
Corresponding author
sergio.cardiaca@gmail.com

Tel.: 55 11 3505-5262
, Marcelo Luiz Peixoto SobralII, Anderson da Silva TerrazasII, Gilmar Geraldo SantosII, Noedir A.G. StolfII,III
I Santa Casa de Itabuna, Cirurgia Cardiovascular, Itabuna/BA, Brazil.
II Hospital Beneficência Portuguesa de São Paulo, Cirurgia Cardiovascular, São Paulo/SP, Brazil.
III Faculdade de Medicina da Universidade de São Paulo, São Paulo/SP, Brazil.
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="cesec10" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cestitle10">INTRODUCTION</span><p id="para10" class="elsevierStylePara elsevierViewall">At the beginning of the twentieth century&#44; Carrell and Guthrie were the first to use homografts to reconstruct dilated vessels&#44; starting a new era in aorta surgery&#46;<a class="elsevierStyleCrossRef" href="#bib1">1</a> In 1952&#44; Cooley and DeBakey conducted the first successful ascending aorta intervention without cardiopulmonary bypass in the resection of a sacciform aneurysm by aortorrhaphy&#46;<a class="elsevierStyleCrossRef" href="#bib2">2</a> In 1956&#44; the same authors performed the first successful replacement of the ascending aorta using cardiopulmonary bypass&#46;<a class="elsevierStyleCrossRef" href="#bib3">3</a> Since then&#44; aortic surgery has developed quickly because of advances in cardiopulmonary bypass&#44; postoperative care&#44; and surgical techniques that reduce the mortality rates of these procedures&#46;<a class="elsevierStyleCrossRefs" href="#bib4">4&#44;5</a> At present&#44; less invasive techniques&#44; such as endovascular interventions&#44; are performed&#44; especially in thoracic and thoracoabdominal aortic disease&#44; depending on factors such as whether the patients would be at high risk during conventional procedures&#46;<a class="elsevierStyleCrossRefs" href="#bib6">6&#44;7</a> Recent studies and case reports have shown the possibility of performing endovascular procedures in the ascending aorta&#44; which seems to be a promising approach&#46;<a class="elsevierStyleCrossRef" href="#bib7">7</a></p><p id="para20" class="elsevierStylePara elsevierViewall">Concerning ascending aorta aneurysms&#44; there are several surgical approaches depending on the level of the disease of the aortic valve&#44; aortic root&#44; sinotubular junction and the ascending aorta&#46; These issues are relevant in the context of congenital diseases such as Marfa&#324;s syndrome&#44;<a class="elsevierStyleCrossRef" href="#bib7">7</a> Ehlers-Danlos syndrome&#44;<a class="elsevierStyleCrossRef" href="#bib8">8</a> congenital aortic valve malformations&#44; and acquired aortic valve diseases&#46;<a class="elsevierStyleCrossRef" href="#bib9">9</a></p><p id="para30" class="elsevierStylePara elsevierViewall">It is accepted that apoptosis is the major mechanism for the control of cell density in developing physiological and pathological conditions affecting smooth muscle cells&#46; It was shown that death signals may be triggered outside the cells by cytokine pathways and stress mechanical forces&#46;<a class="elsevierStyleCrossRef" href="#bib8">8</a> Today&#44; it is known that when there is aortic dilation&#44; there is also cystic medial necrosis&#46; This histological abnormality is characterized by a triad of noninflammatory smooth muscle cell loss&#44; the fragmentation of elastic fibers and the accumulation of basophilic ground substance within cell-depleted areas of the medial layer of the vessel wall&#44; all of which are noninflammatory in nature&#46;<a class="elsevierStyleCrossRef" href="#bib9">9</a></p><p id="para40" class="elsevierStylePara elsevierViewall">Medial degeneration does not uniformly involve the ascending aorta&#46;<a class="elsevierStyleCrossRefs" href="#bib8">8&#44;9</a> The convexity of the vessel looks more damaged because of more severe medial necrosis&#44; greater loss of smooth muscle cells and apoptosis and greater elastic fiber fragmentation&#46; These alterations play an important role in the development of aortic aneurysms because they participate in the matrix remodeling process resulting in the synthesis of extracellular proteins such as collagen&#44; elastin&#44; and proteoglycans&#46;<a class="elsevierStyleCrossRef" href="#bib8">8</a></p></span><span id="cesec20" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cestitle20">CASE REPORT</span><p id="para50" class="elsevierStylePara elsevierViewall">We report the case of a 24-year-old man who presented progressive precordial pain and dyspnea that had worsened in the last six months prior to the consultation with no previous history of arterial hypertension&#44; dyslipidemia&#44; smoking habits&#44; or infectious diseases&#44; such as syphilis or immunodeficiency syndrome&#46; The physical examination showed no signs of congenital syndromes and revealed only severe systolic murmur in the aortic focus&#46; Preoperative examinations were conducted as routine&#46; Laboratory tests as well as the chest radiograph were unchanged&#46; The electrocardiogram showed sinus rhythm&#44; incomplete right bundle branch block and moderate left ventricular hypertrophy&#46; The echocardiogram revealed aortic stenosis caused by a thickened aortic valve&#44; which determined a gradient of 65 mmHg&#44; left ventricular ejection fraction of 0&#46;63&#44; ascending aortic diameter of 30 mm&#44; and systolic and diastolic diameters of 32 and 48 mm&#44; respectively&#46; Then&#44; we proposed the surgery for aortic valve replacement&#46; The procedure was performed by median sternotomy&#46; After the pericardiotomy&#44; we noticed an uncommon aspect of the ascending aorta&#44; with an irregular surface and thinned dilated areas with soft superficial texture &#40;<a class="elsevierStyleCrossRef" href="#fig1">Figure 1</a>&#41;&#46; Aortic cannulation was performed above and medial to the innominate artery&#44; and right atrial cannulation was performed through the right atrial appendage&#46; A left ventricular vent was placed via the right superior pulmonary vein&#46; After total heparinization&#44; cardiopulmonary bypass was then established&#44; the aorta was cross-clamped and a transverse aortotomy was performed&#46; The internal aspect of the vessel was thickened&#44; with multiple focal cavities of different sizes corresponding to the external dilatations&#59; no thrombi were found &#40;<a class="elsevierStyleCrossRef" href="#fig2">Figure 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig1"></elsevierMultimedia><elsevierMultimedia ident="fig2"></elsevierMultimedia><p id="para60" class="elsevierStylePara elsevierViewall">Antegrade cold blood cardioplegic solution was infused every twenty minutes&#46; The three thickened aortic leaflets&#44; as well as the anterolateral convexity and anteromedial concavity of the aorta were excised&#44; leaving the posterior one third&#44; macroscopically normal&#44; to serve as support tissue to the vascular prosthesis that would be placed&#46; Samples were submitted for histological study&#44; and the procedure was completed&#46; The prosthesis implanted was a mechanical no&#46; 21&#59; a Dacron tube prosthesis &#40;no&#46; 24&#41; was positioned in replacement of the ascending aorta above the coronary ostia&#46; De-airing was performed through a catheter placed in the tube&#59; the aortic cross-clamp was removed&#46; As the heartbeats became effective&#44; the cardiopulmonary bypass was ended&#44; and the surgery was finalized as usual&#46; The patient was sent to the intensive care unit&#44; where he remained for one day&#46; He was then discharged by the tenth day in good health&#46; He recovered and has lived until the present moment without complications&#46;</p><p id="para70" class="elsevierStylePara elsevierViewall">The histopathological findings were ascending aorta with areas of intimal fibrosis with dystrophic calcification consistent with arteriosclerosis&#46; Areas of medial cystic degeneration with saccular dilations were observed to be diffusely distributed&#59; inflammation was absent &#40;<a class="elsevierStyleCrossRef" href="#fig3">Figure 3</a>-A&#44;B&#44;C&#41;&#46; The aortic leaflets were fibrous with myxomatous degeneration and dystrophic calcification&#46; The loss of smooth muscle cells was markedly important&#44; as was the deposition of extracellular matrix following the degeneration&#46;</p><elsevierMultimedia ident="fig3"></elsevierMultimedia><p id="para80" class="elsevierStylePara elsevierViewall">Angiotomography of the aorta was performed post-operatively and showed no other injuries in the aorta&#46; Furthermore&#44; the prosthetic graft in the ascending aorta and the mechanic valve prosthesis in the aortic position were properly positioned &#40;<a class="elsevierStyleCrossRef" href="#fig4">Figure 4</a>&#41;&#46;</p><elsevierMultimedia ident="fig4"></elsevierMultimedia><p id="para90" class="elsevierStylePara elsevierViewall">Some laboratory tests were performed in order to try to identify infectious diseases&#44; such as syphilis and HIV&#44; and autoimmune diseases&#44; such as vasculitis&#44; with no positive results&#46;</p></span><span id="cesec30" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cestitle30">DISCUSSION</span><p id="para100" class="elsevierStylePara elsevierViewall">These related microscopic changes determine the thinness of the aortic wall&#59; however&#44; these changes do not explain why this thinning happened in an irregular fashion&#44; causing cavities rather than the uniform dilation that would be expected&#46; The cited histopathological findings confirm cystic medial degeneration&#44; amply described in the medical literature&#44; as the main alteration in those without defined syndromes that could cause modifications in the connective tissues&#46;</p><p id="para110" class="elsevierStylePara elsevierViewall">Some infectious diseases may cause aneurysms of the ascending aorta&#59; however&#44; in these cases&#44; fibrotic thickening and aortic dilation commonly occur&#46; In this case&#44; postoperative examinations were performed but did not identify diseases such as syphilis or potential causes of autoimmune vasculitis&#46; Poststenotic dilation of the ascending aorta often occurs in chronic cases and displays a uniform enlargement pattern&#46;</p><p id="para120" class="elsevierStylePara elsevierViewall">Unfortunately&#44; we did not have the chance to proceed with immunohistochemical studies of the surgical samples&#44; which limits the determination of this diseas&#233;s etiopathogenesis&#46;</p><p id="para130" class="elsevierStylePara elsevierViewall">We decided to perform an ascending aorta replacement&#44; which was not indicated by classical aneurysm definitions&#46; Although the largest diameter was 30 mm&#44; not more than 1&#46;5 times the size of the normal aorta&#44; failure to intervene could cause further complications in the future&#44; such as greater dilations&#44; thromboembolic events&#44; aortic dissection or aortic rupture&#46;</p></span></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="cenpara10">No potential conflict of interest was reported&#46;</p> <p class="elsevierStyleNotepara" id="cenpara20">Santos Jr SF&#44; surgeon assistant&#44; was responsible for the literature researches and manuscript writing&#46; Sobral MLP&#44; surgeon&#44; was also responsible for the literature researches&#46; Terrazas AS&#44; training surgeon&#44; was responsible for the literature researches&#46; Santos GG&#44; co-chief of surgical team&#44; and literature advisor&#46; Stolf NAG&#44; chief of surgical team&#44; literature advisor&#44; and reviewer&#46;</p>"
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          "en" => "<p id="spara30" class="elsevierStyleSimplePara elsevierViewall">A&#41; Aorta segment on the level of the little saccular dilations on the aneurysmatic wall showing a severe lack of smooth muscle cells &#40;Tricromic of Masson&#44; 50x&#41;&#46; B&#41; &#8220;Proteoglycan lakes&#8221; are related to the areas of widespread loss of elastic fibers &#40;Alcian Blue&#44; 200x&#41;&#46; C&#41; Intense elastic fiber fragmentation area &#40;Verhoeff&#44; 200x&#41;&#46;</p>"
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es en pt

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