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Marfan syndrome&#44; VATER syndrome and Palliester&#8211;Killian syndrome&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Although most cases are asymptomatic&#44; patients with partial absence of the pericardium &#40;PAoP&#41; may experience chest pain&#44; palpitations&#44; syncope or even sudden death secondary to herniation of cardiac structures through the pericardial defect&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">We present 2 cases of incidental finding of AoP&#46; The first corresponded to a patient referred at the age of 13 years due to abnormal position of the heart in the chest radiography &#40;CXR&#41;&#46; He was symptomatic and did not have any relevant personal or family history&#46; The physical examination was normal&#46; The electrocardiogram &#40;ECG&#41; &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41; showed sinus bradycardia with right axis deviation &#40;&#43;112 &#176;&#41;&#44; incomplete right bundle branch block &#40;IRBBB&#41;&#44; slow R wave progression&#44; nonspecific intraventricular conduction delay &#40;QRS duration of 96 ms&#41; and abnormal repolarization &#40;inverted T waves in V1&#8211;V4&#44; transitioning in V5 and positive in V6&#41;&#46; The CXR showed lung interposition between the aorta and pulmonary artery &#40;PA&#41;&#44; leftward shift of the heart with no visible right heart border and lung interposition between the left diaphragm and the base of the left heart &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A&#8211;B&#41;&#46; After performing an echocardiogram&#44; PAoP was suspected on account of the posterior and leftward shift of the apex&#44; paradoxical septal motion and apparent dilatation of the right ventricle due to leftward cardiac displacement &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C&#59; Supplemental material Appendix B&#44; videos 1 and 2&#41;&#46; Computed tomography &#40;CT&#41; and magnetic resonance imaging &#40;MRI&#41; findings confirmed the diagnosis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>C&#8211;E&#41;&#46; His exercise test was normal&#46; As the patient was asymptomatic&#44; a conservative approach was chosen&#46; He has remained asmptomatic with no complications after 7 years follow-up&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The second patient was referred at the age of 5 years due to deviation of the heart in the CXR&#46; During surveillance&#44; she was diagnosed with Behcet disease&#46; She was asymptomatic from the cardiac point of view and she did not have any other relevant personal or family history&#46; She had normal physical examination&#46; The ECG &#40;at the age of 15 years&#44; <a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41; revealed mild right axis deviation &#40;&#43;94&#176;&#41;&#44; IRBBB&#44; poor R wave progression&#44; nonspecific intraventricular conduction delay &#40;QRS duration of 98 ms&#41; and abnormal repolarization &#40;inverted T waves in V1&#8211;V3&#44; transitioning in V4 and positive in V5&#8211;V6&#41;&#46; The CXR and echocardiogram &#40;supplementary material Appendix B&#44; videos 3 and 4&#41; revealed the same features described in patient 1&#46; Her diagnosis was confirmed by MRI&#46; She had a normal exercise test&#46; A conservative management was chosen in view of the absence of symptoms&#46; She has remained asymptomatic with no evidence of complications after 13 years of follow-up&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Although AoP is a rare disease&#44; it is important to keep a high level of suspicion&#44; mainly because patients with PAoP&#44; in addition to being symptomatic more frequently&#44; are at risk of herniation and strangulation of the left ventricle through the pericardial defect and&#44; therefore&#44; of sudden death&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3&#44;5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">CXR&#44; ECG&#44; echocardiogram and CT are useful for diagnosis of AoP&#46; However&#44; the gold standard is MRI&#44; which in black blood fast spin echo and steady-state free precession sequences evinces cardiac indentation at the location of the defect&#44; interposition of lung tissue between the aorta and the PA or between the diaphragm and the base of the heart and leftward and posterior displacement of the heart&#46; A rotation angle greater than 60&#176; between the anterior-posterior line passing through the vertebral body and left ventricle main axis in the end-diastolic phase of the axial cine stack offers a good sensitivity for detection of left ventricular AoP&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Although there is no global consensus on the management of these patients&#44; close follow-up and monitoring for complications is recommended in asymptomatic patients in whom AoP is an incidental finding and in the case of complete bilateral or left-sided AoP&#46; Surgery is reserved for patients with PAoP or at risk of complications&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;5</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">In conclusion&#44; we have presented 2 cases of incidental diagnosis of AoP&#44; a disease that is difficult to diagnose due to its rarity and usually asymptomatic&#44; whose early detection is vital due to its potential association with sudden death&#46; Although MRI is the gold standard of diagnosis&#44; the abovementioned imaging investigations can also be useful&#46; Despite the absence of a global consensus&#44; the approach to treatment is conservative&#44; reserving surgery for patients with symptoms or with PAoP at risk of complications&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0040" class="elsevierStylePara elsevierViewall">This research did not receive any external funding&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflicts of interest</span><p id="par0045" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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Scientific Letter
Partial absence of the pericardium: Only an incidental finding?
Ausencia parcial de pericardio: ¿únicamente un hallazgo incidental?
Laia Brunet-Garciaa,
Corresponding author
, Juan Manuel Carretero Bellonb,c, Alex Pérez-Casaresb, Fredy Hermógenes Prada Martínezb, Joan Sánchez-de-Toledob
a Unidad de Cardiología Pediátrica, Consorci Sanitari del Maresme, Hospital de Mataró, Mataró, Barcelona, Spain
b Unidad de Cardiología Pediátrica, Hospital Sant Joan de Déu, Esplugues de Llobregat, Barcelona, Spain
c Unidad de Cardiología Pediátrica, Hospital Universitari Joan XXIII, Universitat Rovira i Virgili, Tarragona, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Absence of the pericardium &#40;AoP&#41; is a rare anomaly usually detected by chance&#46; Its incidence is less than 1 case per 10 000 inhabitants&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is classified based on its extension &#40;complete or partial&#41; and position &#40;left&#44; right or bilateral&#41;&#44; and complete left-sided defects are the most frequent form &#40;70&#37;&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#8211;4</span></a> In 30&#37;&#8211;50&#37; of cases&#44; AoP is associated with congenital heart defects such as tetralogy of Fallot&#44; atrial septal defect&#44; patent ductus arteriosus&#44; mitral valve defects or sinus venosus defects with partial anomalous pulmonary venous drainage<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> as well as pulmonary malformations&#44; Marfan syndrome&#44; VATER syndrome and Palliester&#8211;Killian syndrome&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Although most cases are asymptomatic&#44; patients with partial absence of the pericardium &#40;PAoP&#41; may experience chest pain&#44; palpitations&#44; syncope or even sudden death secondary to herniation of cardiac structures through the pericardial defect&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">We present 2 cases of incidental finding of AoP&#46; The first corresponded to a patient referred at the age of 13 years due to abnormal position of the heart in the chest radiography &#40;CXR&#41;&#46; He was symptomatic and did not have any relevant personal or family history&#46; The physical examination was normal&#46; The electrocardiogram &#40;ECG&#41; &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41; showed sinus bradycardia with right axis deviation &#40;&#43;112 &#176;&#41;&#44; incomplete right bundle branch block &#40;IRBBB&#41;&#44; slow R wave progression&#44; nonspecific intraventricular conduction delay &#40;QRS duration of 96 ms&#41; and abnormal repolarization &#40;inverted T waves in V1&#8211;V4&#44; transitioning in V5 and positive in V6&#41;&#46; The CXR showed lung interposition between the aorta and pulmonary artery &#40;PA&#41;&#44; leftward shift of the heart with no visible right heart border and lung interposition between the left diaphragm and the base of the left heart &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A&#8211;B&#41;&#46; After performing an echocardiogram&#44; PAoP was suspected on account of the posterior and leftward shift of the apex&#44; paradoxical septal motion and apparent dilatation of the right ventricle due to leftward cardiac displacement &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C&#59; Supplemental material Appendix B&#44; videos 1 and 2&#41;&#46; Computed tomography &#40;CT&#41; and magnetic resonance imaging &#40;MRI&#41; findings confirmed the diagnosis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>C&#8211;E&#41;&#46; His exercise test was normal&#46; As the patient was asymptomatic&#44; a conservative approach was chosen&#46; He has remained asmptomatic with no complications after 7 years follow-up&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The second patient was referred at the age of 5 years due to deviation of the heart in the CXR&#46; During surveillance&#44; she was diagnosed with Behcet disease&#46; She was asymptomatic from the cardiac point of view and she did not have any other relevant personal or family history&#46; She had normal physical examination&#46; The ECG &#40;at the age of 15 years&#44; <a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41; revealed mild right axis deviation &#40;&#43;94&#176;&#41;&#44; IRBBB&#44; poor R wave progression&#44; nonspecific intraventricular conduction delay &#40;QRS duration of 98 ms&#41; and abnormal repolarization &#40;inverted T waves in V1&#8211;V3&#44; transitioning in V4 and positive in V5&#8211;V6&#41;&#46; The CXR and echocardiogram &#40;supplementary material Appendix B&#44; videos 3 and 4&#41; revealed the same features described in patient 1&#46; Her diagnosis was confirmed by MRI&#46; She had a normal exercise test&#46; A conservative management was chosen in view of the absence of symptoms&#46; She has remained asymptomatic with no evidence of complications after 13 years of follow-up&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Although AoP is a rare disease&#44; it is important to keep a high level of suspicion&#44; mainly because patients with PAoP&#44; in addition to being symptomatic more frequently&#44; are at risk of herniation and strangulation of the left ventricle through the pericardial defect and&#44; therefore&#44; of sudden death&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3&#44;5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">CXR&#44; ECG&#44; echocardiogram and CT are useful for diagnosis of AoP&#46; However&#44; the gold standard is MRI&#44; which in black blood fast spin echo and steady-state free precession sequences evinces cardiac indentation at the location of the defect&#44; interposition of lung tissue between the aorta and the PA or between the diaphragm and the base of the heart and leftward and posterior displacement of the heart&#46; A rotation angle greater than 60&#176; between the anterior-posterior line passing through the vertebral body and left ventricle main axis in the end-diastolic phase of the axial cine stack offers a good sensitivity for detection of left ventricular AoP&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Although there is no global consensus on the management of these patients&#44; close follow-up and monitoring for complications is recommended in asymptomatic patients in whom AoP is an incidental finding and in the case of complete bilateral or left-sided AoP&#46; Surgery is reserved for patients with PAoP or at risk of complications&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;5</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">In conclusion&#44; we have presented 2 cases of incidental diagnosis of AoP&#44; a disease that is difficult to diagnose due to its rarity and usually asymptomatic&#44; whose early detection is vital due to its potential association with sudden death&#46; Although MRI is the gold standard of diagnosis&#44; the abovementioned imaging investigations can also be useful&#46; Despite the absence of a global consensus&#44; the approach to treatment is conservative&#44; reserving surgery for patients with symptoms or with PAoP at risk of complications&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0040" class="elsevierStylePara elsevierViewall">This research did not receive any external funding&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflicts of interest</span><p id="par0045" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">&#40;A&#41; Resting 12-lead ECG&#44; patient 1&#46; &#40;B&#41; Resting 12-lead ECG&#44; patient 2&#46; &#40;C&#41; Echocardiogram&#44; patient 1&#46; Long axis view showing posterior and rightward deviation of the apex of the left ventricle&#46;</p>"
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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">&#40;A&#8211;C&#41; Chest radiography&#44; posteroanterior &#40;A&#41; and lateral &#40;B&#41;&#59; cardiac CT &#40;C&#41;&#59; leftward shift of the heart with lung interposition between the aorta and the pulmonary artery &#40;&#42;&#41; and interposition of lung parenchyma between the left diaphragm and the border of the base of the left heart &#40;arrow&#41;&#46; &#40;D&#41; Cardiac MRI with the patient in the right lateral decubitus position&#46; &#40;E&#41; Cardiac MRI with patient in supine position showing posterior shift of the cardiac silhouette &#40;arrow&#41;&#46;</p>"
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Article information
ISSN: 23412879
Original language: English
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Anales de Pediatría (English Edition)