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The patient had a history of chronic obstructive pulmonary disease &#40;COPD&#41;&#44; Global Initiative for Chronic Obstructive Lung Disease &#40;GOLD&#41; staging system grade 3&#44; high blood pressure&#44; chronic atrial fibrillation &#40;CAF&#41;&#44; chronic right heart failure &#40;HF&#41; classified as New York Heart Association &#40;NYHA&#41; functional class II&#44; and a 60 pack-year history of cigarette smoking&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Reviewing the patient&#39;s medical record we found a chest radiograph from 1992 &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>&#44; left&#41;&#44; on which mediastinal enlargement was already present&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">During the first decade of follow-up the patient remained stable under standard medical treatment for COPD and HF&#44; and anticoagulation for CAF&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">In 2000 the patient had a gastrointestinal hemorrhage forcing the suspension of anticoagulation&#46; Pharmacological rate control with digoxin was started for treatment of CAF&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">In February 2011 a routine transthoracic echocardiogram revealed moderate to severe dilatation of both atria and mild dilatation of both ventricles with normal thickness of the ventricular walls&#46; There was no significant morphological alteration in valve structures&#46; Moderate mitral regurgitation&#44; mild aortic and pulmonary regurgitation&#44; and mild to moderate tricuspid regurgitation were observed&#46; Pulmonary artery systolic pressure &#40;PASP&#41; was 60 mmHg&#46; Biventricular systolic function was preserved&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">On the basis of the echocardiographic findings&#44; pulmonary hypertension due to lung disease &#40;GOLD grade 3&#41; was diagnosed&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">A routine thoracic computed tomography &#40;CT&#41; scan &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>&#41; was also performed in February 2011 and revealed pronounced dilatation of the pulmonary trunk&#44; with a maximum diameter of 72 mm&#44; and dilatation of the main pulmonary arteries&#44; 40 mm in diameter on the right and 33 mm in diameter on the left&#46; There was no evidence of thrombus in the central pulmonary arteries&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0055" class="elsevierStylePara elsevierViewall">The imaging exams led to a diagnosis of a giant pulmonary artery aneurysm &#40;PAA&#41;&#46; Closer examination of the recent chest radiographs &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>&#44; right&#41; confirmed the persistence of mediastinal enlargement&#44; without significant increase&#44; which&#44; in comparison with the oldest chest radiograph&#44; showed that the evolution of the PAA had been stable for the previous 20 years&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Despite the patient&#39;s stable evolution under medical treatment for the last 10 years&#44; he was admitted to our hospital in March 2012 with severe right HF&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">On admission&#44; physical examination revealed blood pressure of 112&#47;63 mmHg&#44; mild peripheral cyanosis&#44; and increased expiratory time&#44; jugular venous distention&#44; and crackles in both lung bases on pulmonary auscultation&#59; cardiac auscultation revealed arrhythmia with a mean ventricular rate of 70 bpm&#44; and severe lower limb edema was observed&#46;</p><p id="par0070" class="elsevierStylePara elsevierViewall">Laboratory tests performed in the emergency department showed leukocyte count of 4100&#47;&#956;l&#44; 59&#46;7&#37; neutrophils&#44; 12&#46;2 g&#47;dl hemoglobin&#44; and C-reactive protein 5&#46;6 mg&#47;l&#46; On biochemical analysis&#44; electrolytes&#44; renal and liver function&#44; cardiac enzymes and coagulation were all within normal limits&#44; but serum brain natriuretic peptide &#40;BNP&#41; level was elevated &#40;577&#46;4 pg&#47;ml&#41;&#46;</p><p id="par0075" class="elsevierStylePara elsevierViewall">Electrocardiography showed atrial fibrillation with a ventricular rhythm of 63&#47;min&#44; and no significant ST-T abnormalities &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Figure 3</a>&#41;&#46; The chest radiograph &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>&#41; showed an increased cardiothoracic index&#44; mainly due to enlargement of the right atrium and pulmonary artery&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0080" class="elsevierStylePara elsevierViewall">Thoracic CT angiography revealed a PAA 72 mm in diameter but no signs of thromboembolism or thrombosis in the main pulmonary arteries and branches &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Figure 4</a>&#41;&#46;</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0085" class="elsevierStylePara elsevierViewall">The transthoracic echocardiogram showed a PAA &#40;not quantified because of a poor acoustic window&#41;&#44; mild tricuspid regurgitation and PASP of 52 mmHg&#46; The right atrial chamber was dilated and right ventricular systolic function was preserved&#46;</p><p id="par0090" class="elsevierStylePara elsevierViewall">The patient was admitted to the medical ward with a diagnosis of HF and was started on high-dose diuretics&#44; with significant clinical improvement&#46;</p><p id="par0095" class="elsevierStylePara elsevierViewall">A history of major thoracic trauma or significant clinical infection was ruled out&#44; and antinuclear autoantibodies&#44; serology for HIV1 and 2&#44; and the Venereal Disease Research Laboratory and <span class="elsevierStyleItalic">Treponema pallidum</span> hemagglutination tests for syphilis were negative&#46;</p><p id="par0100" class="elsevierStylePara elsevierViewall">After optimization of medical therapy the patient underwent right cardiac catheterization&#44; which revealed mean pulmonary arterial pressure of 26 mmHg&#44; right atrial pressure of 8 mmHg&#44; pulmonary capillary wedge pressure of 12 mmHg&#44; pulmonary vascular resistance of 3&#46;8 Wood units and cardiac index of 2&#46;0 l&#47;min&#47;m<span class="elsevierStyleSup">2</span>&#44; which is compatible with mild precapillary pulmonary hypertension&#46;</p><p id="par0105" class="elsevierStylePara elsevierViewall">The diagnosis was of a giant PAA with precapillary pulmonary hypertension&#44; possibly related to COPD &#40;GOLD grade 3&#41;&#46; Since there is no specific evidence-based treatment for this condition&#44; the patient was discharged on standard HF medication&#44; with the indication for clinical surveillance and specific treatment for COPD&#46; At two-year follow-up he was in a stable condition and the control thoracic CT scan showed no increase in the size of the PAA&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0110" class="elsevierStylePara elsevierViewall">In the literature&#44; several etiologies have been described in the pathogenesis of PAA&#44; including congenital cardiac malformations with intracardiac shunt&#44; vascular abnormalities such as arteritis &#40;e&#46;g&#46;&#44; Takayasu disease&#41; and primary pulmonary hypertension&#44; vasculitis &#40;e&#46;g&#46;&#44; Beh&#231;et&#39;s disease&#41;&#44; connective tissue disorders &#40;such as Marfan&#44; Ehlers-Danlos and Hughes-Stovin syndromes and systemic lupus erythematosus&#41;&#44; tuberous sclerosis&#44; atherosclerosis&#44; hereditary hemorrhagic telangiectasia &#40;Osler-Weber-Rendu disease&#41;&#44; vascular trauma&#44; tuberculosis&#44; syphilis&#44; and bacterial&#44; fungal or other infections&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">5</span></a></p><p id="par0115" class="elsevierStylePara elsevierViewall">Since the introduction of antibiotics&#44; non-infectious causes of PAA have become more common&#44;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">5</span></a> with recent studies revealing an association in more than 50&#37; of cases with congenital heart disease&#46;<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">6</span></a></p><p id="par0120" class="elsevierStylePara elsevierViewall">PAA is defined by pulmonary artery dilatation of &#62;40 mm&#44;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">1</span></a> with some authors considering that dilatation &#62;50 mm defines a giant aneurysm&#46;<a class="elsevierStyleCrossRef" href="#bib0135"><span class="elsevierStyleSup">7</span></a></p><p id="par0125" class="elsevierStylePara elsevierViewall">The clinical manifestations of PAA are diverse and non-specific&#46; Symptoms include chest pain&#44; dyspnea&#44; cough&#44; hemoptysis or palpitations&#46;<a class="elsevierStyleCrossRefs" href="#bib0105"><span class="elsevierStyleSup">1&#44;8</span></a></p><p id="par0130" class="elsevierStylePara elsevierViewall">Nevertheless&#44; it can be asymptomatic&#44; only found incidentally on a routine imaging study such as a chest radiograph&#44; as in our patient&#46;<a class="elsevierStyleCrossRefs" href="#bib0110"><span class="elsevierStyleSup">2&#44;3</span></a></p><p id="par0135" class="elsevierStylePara elsevierViewall">Imaging studies such as CT&#44; echocardiography and cardiac catheterization aid in the diagnosis and prediction of complications&#44; however the gold standard for establishing a diagnosis of PAA is pulmonary angiography&#46;<a class="elsevierStyleCrossRefs" href="#bib0150"><span class="elsevierStyleSup">10&#44;11</span></a></p><p id="par0140" class="elsevierStylePara elsevierViewall">There is no consensus on the management of PAA&#44; as different etiologies lead to different management approaches&#46;<a class="elsevierStyleCrossRef" href="#bib0160"><span class="elsevierStyleSup">12</span></a> In fact&#44; since PAA is a rare lesion&#44; there are no clear guidelines on treatment and patients have been managed individually&#46;<a class="elsevierStyleCrossRef" href="#bib0155"><span class="elsevierStyleSup">11</span></a></p><p id="par0145" class="elsevierStylePara elsevierViewall">Surgical intervention is generally recommended for symptomatic patients and in those with underlying diseases or complications&#44; left-to-right shunt&#44; pulmonary arterial hypertension and large aneurysm size &#40;&#8805;60 mm diameter&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0125"><span class="elsevierStyleSup">5&#44;13&#8211;15</span></a> Nevertheless&#44; some authors consider that PAA&#44; even when very large&#44; confer low risk in the presence of low pulmonary pressures&#44; absence of congenital or acquired significant right shunt and no association with collagenopathies&#46;<a class="elsevierStyleCrossRefs" href="#bib0135"><span class="elsevierStyleSup">7&#44;15</span></a> As these patients have a relatively benign prognosis with an uncomplicated course&#44; some authors&#44; not surprisingly&#44; argue that conservative management is a reasonable strategy&#46;<a class="elsevierStyleCrossRefs" href="#bib0180"><span class="elsevierStyleSup">16&#44;17</span></a></p><p id="par0150" class="elsevierStylePara elsevierViewall">However&#44; little is known about the natural history of PAA&#44; and whatever treatment is chosen&#44; close follow-up is essential&#46;</p><p id="par0155" class="elsevierStylePara elsevierViewall">The approach to high-pressure PAA is even more controversial&#46; Many authors support an aggressive surgical approach in a presence of severe complications such as airway compression&#44;<a class="elsevierStyleCrossRef" href="#bib0190"><span class="elsevierStyleSup">18</span></a> pulmonary artery dissection<a class="elsevierStyleCrossRef" href="#bib0195"><span class="elsevierStyleSup">19</span></a> and intravascular thrombosis&#44; with most reports describing an unfavorable evolution&#44; such as low quality of life or even death&#44; with medical treatment only&#46;<a class="elsevierStyleCrossRefs" href="#bib0120"><span class="elsevierStyleSup">4&#44;9&#44;20</span></a></p><p id="par0160" class="elsevierStylePara elsevierViewall">In the case presented&#44; we describe the longest reported history of an uncomplicated giant high-pressure PAA&#44; with at least 20 years of evolution without significant complications&#44; favoring a conservative approach for such cases&#46;</p><p id="par0165" class="elsevierStylePara elsevierViewall">In our case we suspect that the recent unfavorable evolution of COPD may have contributed to the patient&#39;s HF&#46;</p><p id="par0170" class="elsevierStylePara elsevierViewall">According to the literature&#44; intensification of specific pulmonary hypertension treatment improves airway compression&#46; Maintenance of anticoagulation&#44; although controversial&#44; is usually needed due to the risk of PAA thrombosis&#46; Nevertheless this may be need to be suspended in cases of hemoptysis&#44; progressive growth of the PAA or concomitant dissection&#46;<a class="elsevierStyleCrossRef" href="#bib0200"><span class="elsevierStyleSup">20</span></a> In our case&#44; although indicated&#44; we decided not to start anticoagulation as we believed that the risk of death due to rupture of the PAA in a patient under anticoagulation was higher than the risk of thrombus formation&#46;</p><p id="par0175" class="elsevierStylePara elsevierViewall">Treatment with conventional vasodilators is not recommended in this case since they may impair gas exchange due to the inhibition of hypoxic pulmonary vasoconstriction&#46; In addition&#44; loss of efficacy is observed after long-term use&#46;</p><p id="par0180" class="elsevierStylePara elsevierViewall">Therefore&#44; as the patient had a stable evolution for more than 20 years and his current clinical condition was being treated medically&#44; we decided to maintain a conservative approach and monitor the other clinical conditions&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0185" class="elsevierStylePara elsevierViewall">In the case presented&#44; the recent onset of HF was explained by precapillary pulmonary hypertension associated with COPD in a patient with a giant high-pressure PAA&#46;</p><p id="par0190" class="elsevierStylePara elsevierViewall">A surgical approach was considered&#44; nevertheless&#44; since our patient has severe lung disease which would have significantly increased the surgical risk&#44; we decided not to follow this approach&#46;</p><p id="par0195" class="elsevierStylePara elsevierViewall">In conclusion&#44; giant high-pressure PAA is a rare condition for which in most cases surgical intervention is recommended&#44; nevertheless&#44; since our patient has had a stable evolution for more than 20 years&#44; contradicting the accepted belief found in the literature&#44; we decided to maintain a conservative approach and monitor the patient&#39;s comorbidities closely&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Conflicts of interest</span><p id="par0200" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">The authors report the case of a 74-year-old man&#44; with a history of chronic obstructive pulmonary disease &#40;COPD&#41;&#44; GOLD grade 3&#44; stable for the past two decades&#44; who was admitted to our center with severe right heart failure&#46;</p><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">The chest radiograph showed moderate heart enlargement mainly of the right atrium and pulmonary artery&#44; similar to previous chest radiographs in the previous 20 years&#46; The transthoracic echocardiogram showed a pulmonary artery aneurysm &#40;PAA&#41;&#44; dilatation of the right chambers with pulmonary artery systolic pressure of 52 mmHg&#44; and preserved right ventricular systolic function&#46; A thoracic computed tomography scan confirmed the presence of a giant PAA 72 mm in diameter&#46;</p><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">The patient was started on high-dose diuretics&#44; with significant clinical improvement&#46;</p><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">After optimization of medical therapy right heart catheterization was carried out with the patient in optimal clinical condition&#44; which revealed mild precapillary pulmonary hypertension with a mean pulmonary artery pressure of 26 mmHg&#46;</p><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">On the basis of the clinical and imaging findings a stable&#44; giant&#44; high-pressure&#44; PAA was diagnosed secondary to pulmonary hypertension induced by COPD&#44; with a 20-year follow-up without need for surgical repair&#44; which helped in our decision to maintain medical surveillance&#46; The recent onset of heart failure is explained by the unfavorable evolution of COPD&#46;</p><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">This case may change the attitude expressed in previous studies favoring the choice of an invasive approach to treat giant high-pressure PAAs&#44; instead supporting the maintenance of medical treatment&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Os autores relatam o caso de um homem de 74 anos&#44; com hist&#243;ria de doen&#231;a obstrutiva pulmonar cr&#243;nica &#40;DPOC&#41;&#44; categoria C de GOLD&#44; est&#225;vel nas &#250;ltimas duas d&#233;cadas&#44; admitido por insufici&#234;ncia card&#237;aca grave de c&#226;maras direitas&#46;</p><p id="spar0060" class="elsevierStyleSimplePara elsevierViewall">Na radiografia tor&#225;cica era evidente o alargamento da silhueta card&#237;aca&#44; em particular da aur&#237;cula direita e art&#233;ria pulmonar&#44; sendo sobrepon&#237;vel a radiografias tor&#225;cicas realizadas ao longo dos &#250;ltimos 20 anos&#46; O ecocardiograma transtor&#225;cico documentou um aneurisma da art&#233;ria pulmonar &#40;AAP&#41;&#44; dilata&#231;&#227;o das c&#226;maras direitas&#44; com press&#227;o sist&#243;lica da art&#233;ria pulmonar de 52 mmHg e fun&#231;&#227;o sist&#243;lica do ventr&#237;culo direito preservada&#46; A TC tor&#225;cica confirmou a presen&#231;a de um aneurisma gigante da art&#233;ria pulmonar com 72 mm de di&#226;metro&#46;</p><p id="spar0065" class="elsevierStyleSimplePara elsevierViewall">Iniciou-se terap&#234;utica diur&#233;tica intensiva&#44; com melhoria cl&#237;nica&#46;</p><p id="spar0070" class="elsevierStyleSimplePara elsevierViewall">Ap&#243;s otimiza&#231;&#227;o e com o doente clinicamente est&#225;vel&#44; foi realizado cateterismo card&#237;aco&#44; que revelou hipertens&#227;o pulmonar pr&#233;-capilar ligeira com uma press&#227;o da art&#233;ria pulmonar m&#233;dia de 26 mmHg&#46;</p><p id="spar0075" class="elsevierStyleSimplePara elsevierViewall">Acreditamos que os dados cl&#237;nicos e imagiol&#243;gicos permitiram diagnosticar um aneurisma gigante da art&#233;ria pulmonar de altas press&#245;es secund&#225;rio a hipertens&#227;o pulmonar em contexto de DPOC&#44; atualmente est&#225;vel e com um seguimento de 20 anos sem necessidade de interven&#231;&#227;o cir&#250;rgica&#44; que fundamentou a decis&#227;o de manter a vigil&#226;ncia m&#233;dica&#44; sendo a instala&#231;&#227;o recente de insufici&#234;ncia card&#237;aca explicada pela progress&#227;o da DPOC&#46;</p><p id="spar0080" class="elsevierStyleSimplePara elsevierViewall">Este caso poder&#225; alterar a ideia defendida em estudos pr&#233;vios&#44; que favorecia a escolha de uma abordagem invasiva no tratamento de aneurismas gigantes da art&#233;ria pulmonar de altas press&#245;es&#44; dando mais robustez &#224; manuten&#231;&#227;o de um tratamento m&#233;dico&#46;</p></span>"
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                      "titulo" => "Aneurysm of the pulmonary artery&#58; review of literature and report of a case"
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                        0 => array:2 [
                          "etal" => false
                          "autores" => array:2 [
                            0 => "R&#46;J&#46; Deterling"
                            1 => "O&#46; Clagett"
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                        "tituloSerie" => "Am Heart J"
                        "fecha" => "1947"
                        "volumen" => "34"
                        "paginaInicial" => "471"
                        "paginaFinal" => "499"
                        "link" => array:1 [
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                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/20266464"
                            "web" => "Medline"
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              "etiqueta" => "2"
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                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Proximal pulmonary artery aneurysms in patients with pulmonary artery hypertension&#58; complicated cases"
                      "autores" => array:1 [
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                          "etal" => true
                          "autores" => array:3 [
                            0 => "M&#46; Sakuma"
                            1 => "J&#46; Demachi"
                            2 => "J&#46; Suzuki"
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                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:1 [
                      "Revista" => array:6 [
                        "tituloSerie" => "Intern Med"
                        "fecha" => "2007"
                        "volumen" => "46"
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Case report
Giant high-pressure pulmonary artery aneurysm in an elderly patient with chronic obstructive pulmonary disease
Aneurisma gigante da artéria pulmonar de altas pressões em idoso com doença obstrutiva pulmonar crónica
Sandra A. Moraisa,
Corresponding author
sandramorais13@hotmail.com

Corresponding author.
, Hugo M. Oliveiraa, José R. de Almeidaa, Eduardo Eirasa, Ana Catarina Silvab, Cristina Gavinac
a Serviço de Medicina Interna, ULSM – Hospital Pedro Hispano, Matosinhos, Portugal
b Serviço de Radiologia, ULSM – Hospital Pedro Hispano, Matosinhos, Portugal
c Serviço de Cardiologia, ULSM – Hospital Pedro Hispano, Matosinhos, Portugal
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Pharmacological rate control with digoxin was started for treatment of CAF&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">In February 2011 a routine transthoracic echocardiogram revealed moderate to severe dilatation of both atria and mild dilatation of both ventricles with normal thickness of the ventricular walls&#46; There was no significant morphological alteration in valve structures&#46; Moderate mitral regurgitation&#44; mild aortic and pulmonary regurgitation&#44; and mild to moderate tricuspid regurgitation were observed&#46; Pulmonary artery systolic pressure &#40;PASP&#41; was 60 mmHg&#46; Biventricular systolic function was preserved&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">On the basis of the echocardiographic findings&#44; pulmonary hypertension due to lung disease &#40;GOLD grade 3&#41; was diagnosed&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">A routine thoracic computed tomography &#40;CT&#41; scan &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>&#41; was also performed in February 2011 and revealed pronounced dilatation of the pulmonary trunk&#44; with a maximum diameter of 72 mm&#44; and dilatation of the main pulmonary arteries&#44; 40 mm in diameter on the right and 33 mm in diameter on the left&#46; There was no evidence of thrombus in the central pulmonary arteries&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0055" class="elsevierStylePara elsevierViewall">The imaging exams led to a diagnosis of a giant pulmonary artery aneurysm &#40;PAA&#41;&#46; Closer examination of the recent chest radiographs &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>&#44; right&#41; confirmed the persistence of mediastinal enlargement&#44; without significant increase&#44; which&#44; in comparison with the oldest chest radiograph&#44; showed that the evolution of the PAA had been stable for the previous 20 years&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Despite the patient&#39;s stable evolution under medical treatment for the last 10 years&#44; he was admitted to our hospital in March 2012 with severe right HF&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">On admission&#44; physical examination revealed blood pressure of 112&#47;63 mmHg&#44; mild peripheral cyanosis&#44; and increased expiratory time&#44; jugular venous distention&#44; and crackles in both lung bases on pulmonary auscultation&#59; cardiac auscultation revealed arrhythmia with a mean ventricular rate of 70 bpm&#44; and severe lower limb edema was observed&#46;</p><p id="par0070" class="elsevierStylePara elsevierViewall">Laboratory tests performed in the emergency department showed leukocyte count of 4100&#47;&#956;l&#44; 59&#46;7&#37; neutrophils&#44; 12&#46;2 g&#47;dl hemoglobin&#44; and C-reactive protein 5&#46;6 mg&#47;l&#46; On biochemical analysis&#44; electrolytes&#44; renal and liver function&#44; cardiac enzymes and coagulation were all within normal limits&#44; but serum brain natriuretic peptide &#40;BNP&#41; level was elevated &#40;577&#46;4 pg&#47;ml&#41;&#46;</p><p id="par0075" class="elsevierStylePara elsevierViewall">Electrocardiography showed atrial fibrillation with a ventricular rhythm of 63&#47;min&#44; and no significant ST-T abnormalities &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Figure 3</a>&#41;&#46; The chest radiograph &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>&#41; showed an increased cardiothoracic index&#44; mainly due to enlargement of the right atrium and pulmonary artery&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0080" class="elsevierStylePara elsevierViewall">Thoracic CT angiography revealed a PAA 72 mm in diameter but no signs of thromboembolism or thrombosis in the main pulmonary arteries and branches &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Figure 4</a>&#41;&#46;</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0085" class="elsevierStylePara elsevierViewall">The transthoracic echocardiogram showed a PAA &#40;not quantified because of a poor acoustic window&#41;&#44; mild tricuspid regurgitation and PASP of 52 mmHg&#46; The right atrial chamber was dilated and right ventricular systolic function was preserved&#46;</p><p id="par0090" class="elsevierStylePara elsevierViewall">The patient was admitted to the medical ward with a diagnosis of HF and was started on high-dose diuretics&#44; with significant clinical improvement&#46;</p><p id="par0095" class="elsevierStylePara elsevierViewall">A history of major thoracic trauma or significant clinical infection was ruled out&#44; and antinuclear autoantibodies&#44; serology for HIV1 and 2&#44; and the Venereal Disease Research Laboratory and <span class="elsevierStyleItalic">Treponema pallidum</span> hemagglutination tests for syphilis were negative&#46;</p><p id="par0100" class="elsevierStylePara elsevierViewall">After optimization of medical therapy the patient underwent right cardiac catheterization&#44; which revealed mean pulmonary arterial pressure of 26 mmHg&#44; right atrial pressure of 8 mmHg&#44; pulmonary capillary wedge pressure of 12 mmHg&#44; pulmonary vascular resistance of 3&#46;8 Wood units and cardiac index of 2&#46;0 l&#47;min&#47;m<span class="elsevierStyleSup">2</span>&#44; which is compatible with mild precapillary pulmonary hypertension&#46;</p><p id="par0105" class="elsevierStylePara elsevierViewall">The diagnosis was of a giant PAA with precapillary pulmonary hypertension&#44; possibly related to COPD &#40;GOLD grade 3&#41;&#46; Since there is no specific evidence-based treatment for this condition&#44; the patient was discharged on standard HF medication&#44; with the indication for clinical surveillance and specific treatment for COPD&#46; At two-year follow-up he was in a stable condition and the control thoracic CT scan showed no increase in the size of the PAA&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0110" class="elsevierStylePara elsevierViewall">In the literature&#44; several etiologies have been described in the pathogenesis of PAA&#44; including congenital cardiac malformations with intracardiac shunt&#44; vascular abnormalities such as arteritis &#40;e&#46;g&#46;&#44; Takayasu disease&#41; and primary pulmonary hypertension&#44; vasculitis &#40;e&#46;g&#46;&#44; Beh&#231;et&#39;s disease&#41;&#44; connective tissue disorders &#40;such as Marfan&#44; Ehlers-Danlos and Hughes-Stovin syndromes and systemic lupus erythematosus&#41;&#44; tuberous sclerosis&#44; atherosclerosis&#44; hereditary hemorrhagic telangiectasia &#40;Osler-Weber-Rendu disease&#41;&#44; vascular trauma&#44; tuberculosis&#44; syphilis&#44; and bacterial&#44; fungal or other infections&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">5</span></a></p><p id="par0115" class="elsevierStylePara elsevierViewall">Since the introduction of antibiotics&#44; non-infectious causes of PAA have become more common&#44;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">5</span></a> with recent studies revealing an association in more than 50&#37; of cases with congenital heart disease&#46;<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">6</span></a></p><p id="par0120" class="elsevierStylePara elsevierViewall">PAA is defined by pulmonary artery dilatation of &#62;40 mm&#44;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">1</span></a> with some authors considering that dilatation &#62;50 mm defines a giant aneurysm&#46;<a class="elsevierStyleCrossRef" href="#bib0135"><span class="elsevierStyleSup">7</span></a></p><p id="par0125" class="elsevierStylePara elsevierViewall">The clinical manifestations of PAA are diverse and non-specific&#46; Symptoms include chest pain&#44; dyspnea&#44; cough&#44; hemoptysis or palpitations&#46;<a class="elsevierStyleCrossRefs" href="#bib0105"><span class="elsevierStyleSup">1&#44;8</span></a></p><p id="par0130" class="elsevierStylePara elsevierViewall">Nevertheless&#44; it can be asymptomatic&#44; only found incidentally on a routine imaging study such as a chest radiograph&#44; as in our patient&#46;<a class="elsevierStyleCrossRefs" href="#bib0110"><span class="elsevierStyleSup">2&#44;3</span></a></p><p id="par0135" class="elsevierStylePara elsevierViewall">Imaging studies such as CT&#44; echocardiography and cardiac catheterization aid in the diagnosis and prediction of complications&#44; however the gold standard for establishing a diagnosis of PAA is pulmonary angiography&#46;<a class="elsevierStyleCrossRefs" href="#bib0150"><span class="elsevierStyleSup">10&#44;11</span></a></p><p id="par0140" class="elsevierStylePara elsevierViewall">There is no consensus on the management of PAA&#44; as different etiologies lead to different management approaches&#46;<a class="elsevierStyleCrossRef" href="#bib0160"><span class="elsevierStyleSup">12</span></a> In fact&#44; since PAA is a rare lesion&#44; there are no clear guidelines on treatment and patients have been managed individually&#46;<a class="elsevierStyleCrossRef" href="#bib0155"><span class="elsevierStyleSup">11</span></a></p><p id="par0145" class="elsevierStylePara elsevierViewall">Surgical intervention is generally recommended for symptomatic patients and in those with underlying diseases or complications&#44; left-to-right shunt&#44; pulmonary arterial hypertension and large aneurysm size &#40;&#8805;60 mm diameter&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0125"><span class="elsevierStyleSup">5&#44;13&#8211;15</span></a> Nevertheless&#44; some authors consider that PAA&#44; even when very large&#44; confer low risk in the presence of low pulmonary pressures&#44; absence of congenital or acquired significant right shunt and no association with collagenopathies&#46;<a class="elsevierStyleCrossRefs" href="#bib0135"><span class="elsevierStyleSup">7&#44;15</span></a> As these patients have a relatively benign prognosis with an uncomplicated course&#44; some authors&#44; not surprisingly&#44; argue that conservative management is a reasonable strategy&#46;<a class="elsevierStyleCrossRefs" href="#bib0180"><span class="elsevierStyleSup">16&#44;17</span></a></p><p id="par0150" class="elsevierStylePara elsevierViewall">However&#44; little is known about the natural history of PAA&#44; and whatever treatment is chosen&#44; close follow-up is essential&#46;</p><p id="par0155" class="elsevierStylePara elsevierViewall">The approach to high-pressure PAA is even more controversial&#46; Many authors support an aggressive surgical approach in a presence of severe complications such as airway compression&#44;<a class="elsevierStyleCrossRef" href="#bib0190"><span class="elsevierStyleSup">18</span></a> pulmonary artery dissection<a class="elsevierStyleCrossRef" href="#bib0195"><span class="elsevierStyleSup">19</span></a> and intravascular thrombosis&#44; with most reports describing an unfavorable evolution&#44; such as low quality of life or even death&#44; with medical treatment only&#46;<a class="elsevierStyleCrossRefs" href="#bib0120"><span class="elsevierStyleSup">4&#44;9&#44;20</span></a></p><p id="par0160" class="elsevierStylePara elsevierViewall">In the case presented&#44; we describe the longest reported history of an uncomplicated giant high-pressure PAA&#44; with at least 20 years of evolution without significant complications&#44; favoring a conservative approach for such cases&#46;</p><p id="par0165" class="elsevierStylePara elsevierViewall">In our case we suspect that the recent unfavorable evolution of COPD may have contributed to the patient&#39;s HF&#46;</p><p id="par0170" class="elsevierStylePara elsevierViewall">According to the literature&#44; intensification of specific pulmonary hypertension treatment improves airway compression&#46; Maintenance of anticoagulation&#44; although controversial&#44; is usually needed due to the risk of PAA thrombosis&#46; Nevertheless this may be need to be suspended in cases of hemoptysis&#44; progressive growth of the PAA or concomitant dissection&#46;<a class="elsevierStyleCrossRef" href="#bib0200"><span class="elsevierStyleSup">20</span></a> In our case&#44; although indicated&#44; we decided not to start anticoagulation as we believed that the risk of death due to rupture of the PAA in a patient under anticoagulation was higher than the risk of thrombus formation&#46;</p><p id="par0175" class="elsevierStylePara elsevierViewall">Treatment with conventional vasodilators is not recommended in this case since they may impair gas exchange due to the inhibition of hypoxic pulmonary vasoconstriction&#46; In addition&#44; loss of efficacy is observed after long-term use&#46;</p><p id="par0180" class="elsevierStylePara elsevierViewall">Therefore&#44; as the patient had a stable evolution for more than 20 years and his current clinical condition was being treated medically&#44; we decided to maintain a conservative approach and monitor the other clinical conditions&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0185" class="elsevierStylePara elsevierViewall">In the case presented&#44; the recent onset of HF was explained by precapillary pulmonary hypertension associated with COPD in a patient with a giant high-pressure PAA&#46;</p><p id="par0190" class="elsevierStylePara elsevierViewall">A surgical approach was considered&#44; nevertheless&#44; since our patient has severe lung disease which would have significantly increased the surgical risk&#44; we decided not to follow this approach&#46;</p><p id="par0195" class="elsevierStylePara elsevierViewall">In conclusion&#44; giant high-pressure PAA is a rare condition for which in most cases surgical intervention is recommended&#44; nevertheless&#44; since our patient has had a stable evolution for more than 20 years&#44; contradicting the accepted belief found in the literature&#44; we decided to maintain a conservative approach and monitor the patient&#39;s comorbidities closely&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Conflicts of interest</span><p id="par0200" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">The authors report the case of a 74-year-old man&#44; with a history of chronic obstructive pulmonary disease &#40;COPD&#41;&#44; GOLD grade 3&#44; stable for the past two decades&#44; who was admitted to our center with severe right heart failure&#46;</p><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">The chest radiograph showed moderate heart enlargement mainly of the right atrium and pulmonary artery&#44; similar to previous chest radiographs in the previous 20 years&#46; The transthoracic echocardiogram showed a pulmonary artery aneurysm &#40;PAA&#41;&#44; dilatation of the right chambers with pulmonary artery systolic pressure of 52 mmHg&#44; and preserved right ventricular systolic function&#46; A thoracic computed tomography scan confirmed the presence of a giant PAA 72 mm in diameter&#46;</p><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">The patient was started on high-dose diuretics&#44; with significant clinical improvement&#46;</p><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">After optimization of medical therapy right heart catheterization was carried out with the patient in optimal clinical condition&#44; which revealed mild precapillary pulmonary hypertension with a mean pulmonary artery pressure of 26 mmHg&#46;</p><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">On the basis of the clinical and imaging findings a stable&#44; giant&#44; high-pressure&#44; PAA was diagnosed secondary to pulmonary hypertension induced by COPD&#44; with a 20-year follow-up without need for surgical repair&#44; which helped in our decision to maintain medical surveillance&#46; The recent onset of heart failure is explained by the unfavorable evolution of COPD&#46;</p><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">This case may change the attitude expressed in previous studies favoring the choice of an invasive approach to treat giant high-pressure PAAs&#44; instead supporting the maintenance of medical treatment&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Os autores relatam o caso de um homem de 74 anos&#44; com hist&#243;ria de doen&#231;a obstrutiva pulmonar cr&#243;nica &#40;DPOC&#41;&#44; categoria C de GOLD&#44; est&#225;vel nas &#250;ltimas duas d&#233;cadas&#44; admitido por insufici&#234;ncia card&#237;aca grave de c&#226;maras direitas&#46;</p><p id="spar0060" class="elsevierStyleSimplePara elsevierViewall">Na radiografia tor&#225;cica era evidente o alargamento da silhueta card&#237;aca&#44; em particular da aur&#237;cula direita e art&#233;ria pulmonar&#44; sendo sobrepon&#237;vel a radiografias tor&#225;cicas realizadas ao longo dos &#250;ltimos 20 anos&#46; O ecocardiograma transtor&#225;cico documentou um aneurisma da art&#233;ria pulmonar &#40;AAP&#41;&#44; dilata&#231;&#227;o das c&#226;maras direitas&#44; com press&#227;o sist&#243;lica da art&#233;ria pulmonar de 52 mmHg e fun&#231;&#227;o sist&#243;lica do ventr&#237;culo direito preservada&#46; A TC tor&#225;cica confirmou a presen&#231;a de um aneurisma gigante da art&#233;ria pulmonar com 72 mm de di&#226;metro&#46;</p><p id="spar0065" class="elsevierStyleSimplePara elsevierViewall">Iniciou-se terap&#234;utica diur&#233;tica intensiva&#44; com melhoria cl&#237;nica&#46;</p><p id="spar0070" class="elsevierStyleSimplePara elsevierViewall">Ap&#243;s otimiza&#231;&#227;o e com o doente clinicamente est&#225;vel&#44; foi realizado cateterismo card&#237;aco&#44; que revelou hipertens&#227;o pulmonar pr&#233;-capilar ligeira com uma press&#227;o da art&#233;ria pulmonar m&#233;dia de 26 mmHg&#46;</p><p id="spar0075" class="elsevierStyleSimplePara elsevierViewall">Acreditamos que os dados cl&#237;nicos e imagiol&#243;gicos permitiram diagnosticar um aneurisma gigante da art&#233;ria pulmonar de altas press&#245;es secund&#225;rio a hipertens&#227;o pulmonar em contexto de DPOC&#44; atualmente est&#225;vel e com um seguimento de 20 anos sem necessidade de interven&#231;&#227;o cir&#250;rgica&#44; que fundamentou a decis&#227;o de manter a vigil&#226;ncia m&#233;dica&#44; sendo a instala&#231;&#227;o recente de insufici&#234;ncia card&#237;aca explicada pela progress&#227;o da DPOC&#46;</p><p id="spar0080" class="elsevierStyleSimplePara elsevierViewall">Este caso poder&#225; alterar a ideia defendida em estudos pr&#233;vios&#44; que favorecia a escolha de uma abordagem invasiva no tratamento de aneurismas gigantes da art&#233;ria pulmonar de altas press&#245;es&#44; dando mais robustez &#224; manuten&#231;&#227;o de um tratamento m&#233;dico&#46;</p></span>"
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                    0 => array:2 [
                      "titulo" => "Aneurysm of the pulmonary artery&#58; review of literature and report of a case"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:2 [
                            0 => "R&#46;J&#46; Deterling"
                            1 => "O&#46; Clagett"
                          ]
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                      ]
                    ]
                  ]
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                      "Revista" => array:6 [
                        "tituloSerie" => "Am Heart J"
                        "fecha" => "1947"
                        "volumen" => "34"
                        "paginaInicial" => "471"
                        "paginaFinal" => "499"
                        "link" => array:1 [
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                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/20266464"
                            "web" => "Medline"
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              "etiqueta" => "2"
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                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Proximal pulmonary artery aneurysms in patients with pulmonary artery hypertension&#58; complicated cases"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
                          "autores" => array:3 [
                            0 => "M&#46; Sakuma"
                            1 => "J&#46; Demachi"
                            2 => "J&#46; Suzuki"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:1 [
                      "Revista" => array:6 [
                        "tituloSerie" => "Intern Med"
                        "fecha" => "2007"
                        "volumen" => "46"
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                        "link" => array:1 [
                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/17978536"
                            "web" => "Medline"
                          ]
                        ]
                      ]
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                  ]
                ]
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              "etiqueta" => "3"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Asymptomatic giant pulmonary artery aneurysm in an elderly male patient"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:3 [
                            0 => "M&#46; Khalil"
                            1 => "M&#46; Al-Nozha"
                            2 => "B&#46; Wani"
                          ]
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Idiomas
Revista Portuguesa de Cardiologia (English edition)
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