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Inverted Left Atrial Appendage Bonded to Atrial Septum
Canadian Journal of Cardiology 29 (2013) 1138.e13e1138.e15 www.onlinecjc.ca
Case Report
Inverted Left Atrial Appendage Bonded to Atrial Septum Cláudia Maria Caldas Moura, MD,a Patrícia Isabel Costa Morais Caldas, MD,a António Manuel Rodrigues-Sousa, MD,b Jose Carlos Areias, PhD,a and Jorge Manuel Santos Almeida, MDb a b
Pediatric Cardiology Department, S. João Hospital, Oporto, Portugal
Cardiothoracic Surgery Department, S. João Hospital, Oporto, Portugal
ABSTRACT
RESUM E
Inverted left atrial appendage (LAA) is a rare surgical complication. Our patient, a boy aged 2 years, was diagnosed with a partial atrioventricular defect with mild regurgitation of the left atrioventricular valve and a large primum atrial septal defect. Direct postoperative transesophageal echocardiography revealed a new left atrial mass attached to the atrial septum, without left ventricle inflow obstruction. Out of concern about the nature of this mass, we chose surgical direct examination. Intraoperatively, we diagnosed it as an inverted LAA accidentally attached to the atrial septum suture line. Awareness of this condition can avoid unnecessary diagnostic and therapeutic procedures.
est une complication L’appendice auriculaire gauche (AAG) inverse de 2 ans, a reçu un chirurgicale rare. Notre patient, un garçon âge e à diagnostic de communication auriculoventriculaire partielle associe gurgitation le gère de la valvule auriculoventriculaire gauche et une re une large communication interauriculaire de type ostium primum. chocardiographie transœsophagienne postope ratoire directe L’e ve le une nouvelle masse auriculaire gauche attache e à la cloison a re interauriculoventriculaire, sans obstruction au remplissage du venrant la nature de cette masse, nous avons tricule gauche. Conside choisi l’examen chirurgical direct. Au cours de l’intervention chi la masse comme e tant un AAG rurgicale, nous avons diagnostique attache par accident à la suture de la cloison interauriculaire. inverse viter un diagnostic et des La connaissance de cette affection peut e rapeutiques inutiles. interventions the
Case Report Our patient, a boy aged 2 years, underwent surgical correction of partial atrioventricular defect. Intraoperative transesophageal echocardiogram (TEE) confirmed the previous diagnosis of a large primum atrial septal defect with an unrestrictive colour flow Doppler from the left atrium to the right atrium. Additionally, we found mild regurgitation of the left atrioventricular (AV) valve component by an anterior leaflet cleft (Video 1 , view video online), with both AV valves in a common annulus. During surgery under cardiopulmonary bypass, the cleft of the left AV valve was partially closed, and the coronary sinus was left draining into the right atrium. The atrial septal defect was patched with autologous pericardium. The intraoperative TEE, performed in the operating theater, revealed a new, 10--7-mm left atrial mass linked
to the atrial septum (Fig. 1; Video 2 , view video online) above the left AV valve without left ventricle inflow obstruction. The mass appeared partially mobile and attached to the atrial septum. The 2-dimensional TEE image showed no anatomic continuity with left side structures. Because of concern over the nature of this mass and since the TEE image was not enlightening, we decided on direct examination by the surgeon. The patient was discovered to have an inverted left atrial appendage (LAA) accidentally attached to the atrial septum suture (Fig. 2). After disconnection from the atrial septum suture line, the inverted LAA was easily everted, resulting in the disappearance of the mass on TEE (Video 3 , view video online).
Received for publication November 14, 2012. Accepted December 30, 2012. Corresponding author: Dr Cláudia Moura, Pediatric Cardiology Department, S. João Hospital, Alameda Professor Hernâni Monteiro, 4200-319 Oporto, Portugal. E-mail: [email protected] See page 1138.e15 for disclosure information.
Discussion Inverted LAA is an unusual complication after cardiac operations. Although the prevalence is unknown, it is probably more frequent than other rare causes of left atrial mass. Some cases previously reported in the literature were misdiagnosed most frequently as tumour or thrombus.1 With increasing use of echocardiography to assess surgical repair both during and after surgery, unusual and undiagnosed
0828-282X/$ - see front matter Ó 2013 Canadian Cardiovascular Society. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.cjca.2012.12.017
1138.e14
Canadian Journal of Cardiology Volume 29 2013
Figure 1. Transesophageal echocardiogram, mild esophageal, 4-chamber, 0 : partially mobile left atrial mass (asterisk) bonded to the atrial septum, above the left atrioventricular valve without left ventricle inflow obstruction. LA, left atrium; RA, right atrium.
complications may became apparent. Spontaneous eversion of an inverted LAA has been documented while the heart is being filled.2 Shorter appendages with a wider base are more likely to become inverted. The most frequent cause of postoperative LAA inversion is the negative pressures created during placement of the left atrial vent introduced during surgery.
Additionally, it is possible that the left appendage may be inverted as part of the de-airing manoeuvres.3 The echocardiographic features of inverted LAA have been consistent and include a newly appearing homogeneous mass in the left atrium, usually freely mobile and prolapsing into the mitral valve.
Figure 2. Operative view: inverted left atrial appendage (asterisk) fixed by the left forceps; the right forceps secures the atrial septum. The arrow indicates the point where the appendage was sewed to the septum.
Moura et al. Inverted LAA Bonded to Atrial Septum
Apart from causing confusion in the diagnosis, inversion of the LAA can be clinically noteworthy. Ankersmit et al.3 believe that inverted LAA should be considered as a thrombogenic entity once a patient is found with a possible embolus associated to a de novo inverted LAA. On the other hand, Allen et al.4 have suggested that the lesion might be left alone because of the nature of the totally endothelialized structure. In our patient inverted LAA was not considered at first once the mass was linked to the atrial septum, and 2-dimensional TEE showed no anatomic continuity with left side structures. The hyperechogenic appearance, near the left atrial appendage, was assigned as the well-developed ligament of Marshall seen in our patient. In our case, spontaneous eversion of the LAA was impossible since it was sewn to the atrial septum suture line. In conclusion, an inverted LAA must be considered as one of the causes of left atrial mass observed during routine perioperative echocardiogram. Intraoperative detection and immediate surgical correction are important in order to avoid unnecessary reoperation. Disclosures The authors have no conflicts of interest to disclose.
1138.e15
References 1. Ankersmit HJ, Mohl W, Moser B, et al. De novo inverted left atrial appendage: an unrecognized cause of left atrial mass with symptoms mimicking myxoma. J Thorac Cardiovasc Surg 2001;121:1211-3. 2. Toma DM, Stewart RB, Miyake-Hull CY, Otto CM. Inverted left atrial appendage mimicking a left atrial mass during mitral valve repair. J Am Soc Echocardiogr 1995;8:557-9. 3. Ankersmit HJ, Kocher A, Frank H, Mohl W, Wolner E. Inverted left atrial appendage masquerading as myxoma. Circulation 2000;101:e42-3. 4. Allen BS, Ilbawi M, Hartz R, Kumar S, Thoele D. Inverted left atrial appendage: an unrecognized cause of left atrial mass. J Thoracic Cardiovasc Surg 1997;114:278-80.
Supplementary Material To access the supplementary material accompanying this article, visit the online version of the Canadian Journal of Cardiology at www.onlinecjc.ca and at http://dx.doi:10.1016/ j.cjca.2012.12.017
Case Report
Inverted Left Atrial Appendage Bonded to Atrial Septum Cláudia Maria Caldas Moura, MD,a Patrícia Isabel Costa Morais Caldas, MD,a António Manuel Rodrigues-Sousa, MD,b Jose Carlos Areias, PhD,a and Jorge Manuel Santos Almeida, MDb a b
Pediatric Cardiology Department, S. João Hospital, Oporto, Portugal
Cardiothoracic Surgery Department, S. João Hospital, Oporto, Portugal
ABSTRACT
RESUM E
Inverted left atrial appendage (LAA) is a rare surgical complication. Our patient, a boy aged 2 years, was diagnosed with a partial atrioventricular defect with mild regurgitation of the left atrioventricular valve and a large primum atrial septal defect. Direct postoperative transesophageal echocardiography revealed a new left atrial mass attached to the atrial septum, without left ventricle inflow obstruction. Out of concern about the nature of this mass, we chose surgical direct examination. Intraoperatively, we diagnosed it as an inverted LAA accidentally attached to the atrial septum suture line. Awareness of this condition can avoid unnecessary diagnostic and therapeutic procedures.
est une complication L’appendice auriculaire gauche (AAG) inverse de 2 ans, a reçu un chirurgicale rare. Notre patient, un garçon âge e à diagnostic de communication auriculoventriculaire partielle associe gurgitation le gère de la valvule auriculoventriculaire gauche et une re une large communication interauriculaire de type ostium primum. chocardiographie transœsophagienne postope ratoire directe L’e ve le une nouvelle masse auriculaire gauche attache e à la cloison a re interauriculoventriculaire, sans obstruction au remplissage du venrant la nature de cette masse, nous avons tricule gauche. Conside choisi l’examen chirurgical direct. Au cours de l’intervention chi la masse comme e tant un AAG rurgicale, nous avons diagnostique attache par accident à la suture de la cloison interauriculaire. inverse viter un diagnostic et des La connaissance de cette affection peut e rapeutiques inutiles. interventions the
Case Report Our patient, a boy aged 2 years, underwent surgical correction of partial atrioventricular defect. Intraoperative transesophageal echocardiogram (TEE) confirmed the previous diagnosis of a large primum atrial septal defect with an unrestrictive colour flow Doppler from the left atrium to the right atrium. Additionally, we found mild regurgitation of the left atrioventricular (AV) valve component by an anterior leaflet cleft (Video 1 , view video online), with both AV valves in a common annulus. During surgery under cardiopulmonary bypass, the cleft of the left AV valve was partially closed, and the coronary sinus was left draining into the right atrium. The atrial septal defect was patched with autologous pericardium. The intraoperative TEE, performed in the operating theater, revealed a new, 10--7-mm left atrial mass linked
to the atrial septum (Fig. 1; Video 2 , view video online) above the left AV valve without left ventricle inflow obstruction. The mass appeared partially mobile and attached to the atrial septum. The 2-dimensional TEE image showed no anatomic continuity with left side structures. Because of concern over the nature of this mass and since the TEE image was not enlightening, we decided on direct examination by the surgeon. The patient was discovered to have an inverted left atrial appendage (LAA) accidentally attached to the atrial septum suture (Fig. 2). After disconnection from the atrial septum suture line, the inverted LAA was easily everted, resulting in the disappearance of the mass on TEE (Video 3 , view video online).
Received for publication November 14, 2012. Accepted December 30, 2012. Corresponding author: Dr Cláudia Moura, Pediatric Cardiology Department, S. João Hospital, Alameda Professor Hernâni Monteiro, 4200-319 Oporto, Portugal. E-mail: [email protected] See page 1138.e15 for disclosure information.
Discussion Inverted LAA is an unusual complication after cardiac operations. Although the prevalence is unknown, it is probably more frequent than other rare causes of left atrial mass. Some cases previously reported in the literature were misdiagnosed most frequently as tumour or thrombus.1 With increasing use of echocardiography to assess surgical repair both during and after surgery, unusual and undiagnosed
0828-282X/$ - see front matter Ó 2013 Canadian Cardiovascular Society. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.cjca.2012.12.017
1138.e14
Canadian Journal of Cardiology Volume 29 2013
Figure 1. Transesophageal echocardiogram, mild esophageal, 4-chamber, 0 : partially mobile left atrial mass (asterisk) bonded to the atrial septum, above the left atrioventricular valve without left ventricle inflow obstruction. LA, left atrium; RA, right atrium.
complications may became apparent. Spontaneous eversion of an inverted LAA has been documented while the heart is being filled.2 Shorter appendages with a wider base are more likely to become inverted. The most frequent cause of postoperative LAA inversion is the negative pressures created during placement of the left atrial vent introduced during surgery.
Additionally, it is possible that the left appendage may be inverted as part of the de-airing manoeuvres.3 The echocardiographic features of inverted LAA have been consistent and include a newly appearing homogeneous mass in the left atrium, usually freely mobile and prolapsing into the mitral valve.
Figure 2. Operative view: inverted left atrial appendage (asterisk) fixed by the left forceps; the right forceps secures the atrial septum. The arrow indicates the point where the appendage was sewed to the septum.
Moura et al. Inverted LAA Bonded to Atrial Septum
Apart from causing confusion in the diagnosis, inversion of the LAA can be clinically noteworthy. Ankersmit et al.3 believe that inverted LAA should be considered as a thrombogenic entity once a patient is found with a possible embolus associated to a de novo inverted LAA. On the other hand, Allen et al.4 have suggested that the lesion might be left alone because of the nature of the totally endothelialized structure. In our patient inverted LAA was not considered at first once the mass was linked to the atrial septum, and 2-dimensional TEE showed no anatomic continuity with left side structures. The hyperechogenic appearance, near the left atrial appendage, was assigned as the well-developed ligament of Marshall seen in our patient. In our case, spontaneous eversion of the LAA was impossible since it was sewn to the atrial septum suture line. In conclusion, an inverted LAA must be considered as one of the causes of left atrial mass observed during routine perioperative echocardiogram. Intraoperative detection and immediate surgical correction are important in order to avoid unnecessary reoperation. Disclosures The authors have no conflicts of interest to disclose.
1138.e15
References 1. Ankersmit HJ, Mohl W, Moser B, et al. De novo inverted left atrial appendage: an unrecognized cause of left atrial mass with symptoms mimicking myxoma. J Thorac Cardiovasc Surg 2001;121:1211-3. 2. Toma DM, Stewart RB, Miyake-Hull CY, Otto CM. Inverted left atrial appendage mimicking a left atrial mass during mitral valve repair. J Am Soc Echocardiogr 1995;8:557-9. 3. Ankersmit HJ, Kocher A, Frank H, Mohl W, Wolner E. Inverted left atrial appendage masquerading as myxoma. Circulation 2000;101:e42-3. 4. Allen BS, Ilbawi M, Hartz R, Kumar S, Thoele D. Inverted left atrial appendage: an unrecognized cause of left atrial mass. J Thoracic Cardiovasc Surg 1997;114:278-80.
Supplementary Material To access the supplementary material accompanying this article, visit the online version of the Canadian Journal of Cardiology at www.onlinecjc.ca and at http://dx.doi:10.1016/ j.cjca.2012.12.017