© 2014, Wiley Periodicals, Inc. DOI: 10.1111/echo.12667

Echocardiography

REVIEW ARTICLE

Left Atrial Appendage Aneurysm: A Systematic Review of 82 Cases Madan Raj Aryal, M.D.,* Fayaz A. Hakim, M.D.,† Sailu Ghimire, M.B.B.S.,‡ Sushil Ghimire, M.B.B.S.,§ Smith Giri, M.D.,¶ Anil Pandit, M.D.,† Yashoda Bhandari, R.N.,** Naresh Bhandari, M.D.,†† Ranjan Pathak, M.D.,* Paras Karmacharya, M.D.,* and Rajesh Pradhan, M.D.‡‡ *Department of Internal Medicine, Reading Health System, West Reading, Pennsylvania; †Division of Cardiovascular Diseases, Department of Medicine, Mayo Clinic, Scottsdale, Arizona; ‡College of Medical Sciences, Kathmandu University, Bharatpur, Nepal; §Department of Internal Medicine, Tribhuvan University Teaching Hospital, Kathmandu, Nepal; ¶Department of Medicine, University of Tennessee Health Science Center Memphis, Memphis, Tennessee; **Department of Nursing, Alvernia University, Reading, Pennsylvania; ††Department of Internal Medicine, St. Agnes Hospital, Baltimore, Maryland; and ‡‡Regional Heart Doctors, Rapid City Regional Hospital, Rapid City, South Dakota

Background: Aneurysm of the left atrial appendage is rare. We sought to systematically review the published literature on left atrial appendage aneurysm (LAAA) to address its demographic features, clinical characteristics, treatment, complications, and outcomes. Methodology: A systematic electronic search of Medline, PubMed, and EMBASE for case reports, case series, and related articles of LAAA published from 1962 until July 2013 was carried out. Statistical analysis was done using SPSS version 20.0. Logistic Regression Analysis was used to identify the independent predictors of LAAA-related thrombus formation and embolism. Results: Eighty-two cases of LAAA were identified. There was a slight female preponderance and most of the patients presented in their third decades. Palpitation, dyspnea or both were most common clinical symptoms associated with LAAA. Echocardiography was the main diagnostic modality used and the mean size of aneurysm was 7.08  3.03 9 5.75  2.36 cm. Surgical resection of the aneurysm was performed in most patients with favorable results. Systemic embolism and atrial tachyarrhythmias were the two common complications associated with untreated LAAA. Presence of atrial fibrillation/flutter was the only significant predictor of thrombus formation/embolic events. Conclusion: Aneurysm of left atrial appendage is rare and often an incidental diagnosis during echocardiography. It is important to recognize this entity since it is associated with cardiovascular morbidity and mortality by predisposing to atrial tachyarrhythmia and thromboembolism. Surgical resection is the standard of treatment in the current literature. Medical management is directed toward the treatment of thromboembolism and atrial tachyarrhythmia. (Echocardiography 2014;00:1–8) Key words: left atrial appendage aneurysm, echocardiogram, aneurysectomy, atrial tachyarrhythmia, thromboebolism

Left atrial appendage aneurysm is a rare acquired or congenital condition1–4 with less than 100 cases reported in the literature. The condition is characterized by localized or diffuse dilatation of left atrial appendage. It is seen in all ages ranging from infancy to ninth decade of life. Patients with left atrial appendage aneurysm (LAAA) may be asymptomatic, or present with dyspnea, palpitations, and/or thromboembolic phenomenon. The condition may be detected incidentally during cardiac imaging. With the increasing use of transthoracic echocardiography Address for Correspondence and reprint requests: Madan Raj Aryal, M.D., Reading Health System, 6th Avenue and Spruce Street, West Reading, Pensylvania 19611. Fax: 484-628-9292; E-mail: [email protected]

(TTE), transesophageal echocardiography (TEE), magnetic resonance imaging (MRI), and computed tomography (CT), LAAA are being recognized more frequently. Surgical resection is indicated for LAAA as it predisposes to thromboembolism and atrial tachycarrhythmias. Search Strategy and Data Collection: A systematic electronic search of Medline, PubMed, and EMBASE for case reports, case series, and related articles of LAAA published from 1962 till July 2013 was performed independently by 3 authors (MRA, SG, and NB) using “left atrium,” “appendage,” “aneurysm,” “cardiac,” “congenital,” “atrial,” and “acquired” in addition to the medical subject heading (MESH) terms “atrial 1

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appendage” and “aneurysm” as search terms. The search was limited to human studies. Bibliographies of the reviewed articles were further scanned to identify additional reports. Care was taken to avoid duplication. Eighty-two cases published in English language were identified. The demographic variables, clinical presentations, diagnostic modalities, treatment, outcome, and complications of LAAA were studied. Statistical analyses were performed using SPSS Statistics for Windows, Version 20.0 (IBM Corp, Armonk, NY, USA). Binomial logistic regression analysis was used to identify independent predictors of LAAArelated thrombus formation/embolism. Results: Demographics and Clinical Presentation: We identified 82 cases of LAAA; 36 were males and 44 were females. The sex in 2 patients was not mentioned. The mean age of presentation was 31  20 years (range: fetus at 28 weeks to 88 years) (Fig. 1). Age prevalence was highest in the third decade of life. Thirty-two (39%) cases were reported to have congenital LAAA. The most common clinical presentations associated with LAAA were palpitation reported in 35 (43%) followed by dyspnea in 18 (22%), arrhythmia in 12 (15%), and systemic thromboembolic phenomenon (stroke) in 9 (11%) patients. Four other cases developed thromboembolic complications later during the course. Thirteen (16%) patients were asymptomatic and diagnosed incidentally. Six (7%) patients reported nonspecific chest pain and 2 had congestive heart failure. The physical examination was documented in 62 cases. Thirteen (16%) patients had systolic murmur, 11 (13%) had irregularly irregular pulse and 10 (12%) patients had tachycardia. Associated congenital anomalies were reported in 5 patients; 3 with atrial septal defect (ASD), 1 with ventricular septal defect (VSD), 1 with

Figure 1. Age distribution of LAAA (n = 82). LAAA = left atrial appendage aneurysm.

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anomaly of the renal artery, and 1 with Noonan syndrome. Investigations: Chest x-ray was done in 76 (93%) patients and prominent/widened/bulged left heart border and cardiomegaly were reported in 36 (47%) and 23 (30%) patients, respectively. Sixty-eight (83%) patients had an electrocardiogram (ECG). The rhythm was documented in 63 cases: Atrial fibrillation/flutter in 33 (52%), supraventricular tachycardia in 2 (3%), and normal sinus rhythm in 28 (44%). Three patients had findings described as broad notched P-wave, pathological Q-wave, and left axis deviation and 2 patients were noted to have conduction defects. Sixty-six (80%) patients underwent TTE and LAAA was diagnosed accurately in 30 (45%). It was described as cyst, cavity, mass, echo free structure, and echolucent space in 34 (38%) and not detected at all in 2 patients. Twentynine patients (35%), including 25 of 29 patients who initially had TTE underwent TEE and LAAA was diagnosed in 26 (90%). In the remaining 3 (10%) patients it was reported either as dilated left atrium or a cystic mass. Mitral regurgitation was reported in 4 (6%) patients during TTE. One patient had an associated ASD, VSD, right atrial appendage aneurysm, and pulmonary hypertension. Thirty patients underwent preoperative cardiac catheterization (in many cases specific type of cardiac catheterization such as left ventriculography or left atrial angiography was not mentioned) and LAAA was diagnosed in 17 (57%) cases. With the improvement in echocardiographic techniques and the availability of TEE, angiography is rarely used in current day practice for diagnosing LAAA. Sixteen patients had CT scan (8 modality not specified, 3 multidetector CT, 2 contrast CT chest, 1 cardiac CT, 1 noncontrast thoracic CT, and 1 3D CT) and LAAA was detected in 11 (69%). Twenty-one (26%) patients also underwent cardiac MRI and LAAA was diagnosed in 17 (80%) cases. Both CT and MRI were performed to further evaluate the abnormalities detected during echocardiography especially in patients in whom the diagnosis was not clear. Intracardiac (left atrial and left ventricular) thrombi were noted in 21(26%) patients. The LAAA size by TTE was highly variable with the smallest measuring 2.2 9 1.1 cm and the largest 13 9 10 cm. The average size of LAAA was 7.08  3.03 9 5.75  2.36 cm. The size at the time of surgery was reported in 25 patients; mean size was 10.25 9 8.03 cm (range 2.9 9 2.2 cm to 22 9 15 cm). Fibrosis of the aneurysmal wall was the most common histological finding reported in 20/32 (63%) patients.

Left Atrial Appendage Aneurysm, Systematic Review

Treatment and prognosis: Of the 76 cases where treatment of the LAAA was mentioned, 64 (84%) patients underwent surgical treatment with either aneurysmectomy in 36 (56%) or excision of the left atrial appendage in 17 (27%). The exact surgical approach (aneurysmectomy versus left atrial appendage excision) was not clear in the remaining 11 (17%) patients. The surgery was performed upon diagnosis of LAAA regardless of patient’s symptoms or the size of aneurysm (most common reason being presence of arrhythmias, thromboembolic complications or prevention of thromboembolic complication, and left ventricular dysfunction due to compression by the LAAA). In patients with intracardiac thrombi, surgical intervention was delayed to allow resolution of thrombi with anticoagulation. One patient was found to be inoperable due to extensive adhesions at the time of surgery. Of the total 27 patients with documented AF, 5 (9%) patients underwent either ablation or maze procedure at the time of LAAA excision. Postoperatively 29 patients were followed up for a median period of 180 days (range 6 weeks to 6 years) with no reported adverse events. Eleven (15%) did not undergo surgery (5 refused surgery,5–9 2 with no arrhythmia or thrombus, 2 reasons not mentioned, 1 autopsy diagnosis, and 1 had bilateral atrial appendage aneurysm and ASD/VSD with Eisenmenger’s syndrome10). These patients were treated conservatively with anticoagulants for either presence of thromboembolic complications or for prophylaxis. Antiarrythmics medications (b-blocker, digoxin and amiodarone) were used for atrial tachyarrhythmias when present. Three patients with thromboembolism were not anticoagulated: patient refusal (1), autopsy diagnosis (1), and reason not mentioned (1). The anticoagulation was discontinued in patients following surgical treatment of LAAA. Stepwise logistic regression analysis was used to identify predictors of LAAA-related thrombus (left atrial or LAAA) formation and embolism. Variables included were age, sex, LAAA area in cm2 (length 9 breadth), AF/flutter, and type of LAAA (congenital or acquired). By multivariate analysis AF/flutter was the only variable significantly associated with thrombus/embolism (P < 0.05). Using logistic regression analysis with enter method, presence of AF/flutter was the only significant predictor of thrombus/embolic event (Table I). Multivariate logistic regression analysis was used to identify predictors of LAAA-related thrombus (left atrial or LAAA) formation and embolism. Variables included were age, sex, LAAA area in cm2 (length 9 breadth), AF/flutter,

TABLE I Binary Logistic Regression Analysis to Identify the Predictors of Thrombus/Thromboembolism in Study Population 95% CI of Odds Ratio

Predictor Age Gender Congenital versus acquired Presence of atrial fibrillation/Flutter Area of aneurysm (cm2)

Lower Level

Upper Level

P-Value

1.00 0.87 0.83

0.97 0.33 0.29

1.03 2.31 2.35

0.80 0.87 0.72

2.7

1.2

7.48

0.04

0.99

0.97

1.00

0.89

Odds Ratio

CI = Confidence Interval; OR = Odds Ratio. P < 0.05 is considered significant.

and type of LAAA (congenital or acquired). This analysis demonstrated that AF/flutter was the only variable significantly associated with thrombus/embolism (P < 0.05), (Table I). Discussion: Historical Perspective: LAAA is characterized by a localized outpouching or a diffuse enlargement of left atrial appendage. However, there is no clear consensus on the definition of the LAAA. The first case of LAAA was reported by Parmley et al. in 1962 during a surgical procedure. Godwin et al. in 1968 attempted to visualize the LAAA with I131-labeled albumin, which till then could be confirmed only by contrast angiography and surgical exploration.11 Bramlet et al. in 1981 reported a case of LAAA diagnosed at autopsy.12 In 1990, Frambach and Comess et al. separately reported 2 cases of LAAA diagnosed during TTE13 and TEE,14 respectively. With routine use of echocardiography LAAA is reported more frequently and has emerged as a disease entity that has been shown to be associated with morbidity and mortality. Epidemiology, Pathogenesis, Gross, and Microscopic Features: In our review of 82 cases, most patients were in their third decade, although LAAA has been reported across all the age groups (Fig. 1).15–17 There was a slight female predominance, which is similar to previous report of 34 patients by Frambach et al.13 Two fifths of the cases of LAAA are congenital. The use of fetal echocardiography has made it possible to diagnose this condition even before the birth.18 Whether congenital or acquired, LAAA grow in size over several years 3

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and tend to become symptomatic with increasing risk of thromboembolism.1,18 Surgical treatment seems to be important in management of LAAA as there was no reported thromboembolic and arrhythmic episodes or any other complications after the resection. However, because of the rarity of this entity, direct comparison of medical treatment with anticoagulation and antiarrhythmic therapy is not available in the current literature. LAAA has been classified as intrapericardial or extrapericardial. The extrapericardial type is associated with pericardial defect through which the appendage can herniate and undergo aneurysmal dilatation with time. On the other hand, intrapericardial type is thought to be due to developmental weakness in the wall of left atrium and or appendage.19,20 However, this distinction can only be made during surgery or at autopsy.21–23 LAAA has also been classified as congenital or acquired. Congenital form arises from dysplasia of musculi pectinati and related atrial muscle bands.21 Acquired forms often occur in association with left atrial enlargement due to mitral valve disease (mitral stenosis and or mitral regurgitation).1,3,4 LAAA has been reported to occur in association with other congenital anomalies such as ASD, VSD, anomalous renal artery, and Noonan syndrome as outlined in this review.10,24–26 Fibrosis of the endocardium or myocardium is a common histopathological finding in a thin walled aneurysm. Myocardial hypertrophy and fatty infiltration of myocardial fibers may be seen is some cases.20,22,27–30

tachycardia may occur due to LAAA.35 The physical examination may be normal or be significant for tachycardia,36 irregularly irregular pulse, systolic murmur,17,37–39 and signs of systemic thromboembolism.6,38,40–42 There is no clear association of these physical findings to LAAA itself, and are likely a manifestation of the underlying cardiovascular disease. The usual findings on chest x-ray are cardiomegaly with a prominent upper left heart border.43–46 ECG and 24-hour Holter monitoring may reveal atrial tachyarrhythmias (Fig. 2)32,47 and atrioventricular or intraventricular conduction defects.7,23,24,48 TTE has a limited value in the diagnosis of LAAA with a sensitivity of 45% and LAAA may be mistaken for or described as a cyst, cavity, mass, echo free structure, echolucent space, and dilated left atrium47,49–54 necessitating additional studies (Fig. 3). TEE (movie clip S1) is superior to TTE for the detection of LAAA, with a sensitivity of 90%. TEE should be performed in patients where there is ambiguity in the diagnosis during TTE examination. TEE is also useful in the detection of intra-atrial and or left atrial appendage thrombi.47,55,56 CT with or without contrast and MRI may be used as an adjunct for better definition of anatomy, studying the relationship of LAAA with neighboring structures and confirming alternate diagnoses and associated congenital anomalies.57,58 Angiocardiography,57–62 and radionuclide scintigraphy,48– 50,63 used in the past are rarely needed for the diagnosis or evaluation of LAAA.

Clinical Feature and Diagnosis: Palpitations and or dyspnea are the most common symptoms associated with LAAA. However, it is hard to discern if these symptoms have a cause–effect relationship with LAAA or are simply a manifestation of some other underlying cardiac disorder. Aneurysmal dilatation of the LAA and/ or enlarged left atrium forms a substrate to triggered or reentrant atrial tachyarrhythmia.31 In addition, associated abnormalities of the conduction system may also predispose to arrhythmia. There is, however, no study proving the origin of the atrial arrhythmias from the LAAA with an electrophysiologic study. Aneurysmal dilatation leads to stasis of blood and subsequent thrombi formation. However, the size of the aneurysm was not found to be predictive of thromboembolism on multivariate analysis. Chest pain due to tension on the pericardium and heart failure leading to shortness of breath has also been reported.3,32–34 Frambach et al. reported a 15year-old boy presenting with chest pain due to temporary compression of the left anterior descending artery by LAAA leading to myocardial ischemia.13 Rarely, supine hypotension and

Treatment and Prognosis: Most of the authors proposed surgery (aneurysmectomy) as the main treatment modality for LAAA regardless of the presence or absence of symptoms.64,65 A median sternotomy approach with

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Figure 2. ECG in a patient with LAAA showing atrial flutter. ECG = electrocardiogram; LAAA = left atrial appendage aneurysm.

Left Atrial Appendage Aneurysm, Systematic Review

Figure 3. Transesophageal echocardiography showing the left atrial appendage aneurysm. Spontaneous echo contrast is seen in the left atrial appendage aneurysm.

or without cardiopulmonary bypass or minimal endoscopic techniques are both acceptable options.22,32,66 More than 83% of the patients in this review underwent surgical treatment. As the manipulation of aneurysm during surgery may dislodge thrombi, cardiopulmonary bypass is instituted if preoperative or intraoperative echocardiogram shows a thrombus or a large LAAA.13 LAAA have been safely resected with minimally invasive endoscopic techniques and should be considered for patients with small LAAA in the absence of left atrial and/or appendage thrombi.52 Surgical or endoscopic resection eliminates the risk of both thromboembolism and tachyarrhythmia. Resection of very large (giant) LAAA may leave dysmorphic atrial tissue that can predispose to atrial tachyarrhythmia. Hence, in patients with large LAAA and those with associated atrial fibrillation and/or biatrial enlargement concurrent Cox-Maze III type or other ablative procedure at the time of LAAA resection should be performed.67–70 In centers that do not perform these procedures, excision of LAAA alone appears to be a reasonable choice.19,21,33,53 The prognosis after surgical treatment is favorable as none of the patients in this review reported recurrent symptoms, arrhythmia, or thromboembolic events at up to 180 days follow-up. In those not undergoing surgery due to various reasons medical management should be directed to treat atrial tachyarrhythmia and thromboembolic complication.7,8,71,72 Limitations of the Study: Several limitations of our study should be noted. This is a retrospective series of reported cases in the current literature, and has inherent biases related to such studies including selection and

publication biases. In this review, we included only the abstracts available in English language. Asymptomatic cases of LAAA are unlikely to be recognized; hence, the reported cases may not represent the overall patient population with LAAA. There is no clear and consensus definition of what constitutes a LAAA. Foale et al. proposed diagnostic criteria for congenital aneurysms of the left atrium that included (1) origin from an otherwise normal atrial chamber, (2) a clearly defined communication with the atrial cavity, and (3) their intrapericardial location with resultant distortion of the left ventricle.73 However, there is no consensus on what size is “aneurismal.” The size of the LAAA as measured by TTE ranged between 2.2 9 1.1 cm and 13 9 10 cm. The smallest LAAA treated surgically was 2.9 9 2.2 cm. There are several reports of “giant LAAA” with the largest reported size of 13 9 10 cm. However, the size of measurement is also highly variable with some authors reporting the maximal width and the depth of the aneurysm on TTE, others reporting 2 axis measurement of the neck of the LAA in TEE, whereas some authors did not report where the measurements were taken. In a study of 500 normal autopsy hearts, Veinot et al. reported the size of normal LAA in different age groups and sexes.74 The mean diameter of the orifice, width of the body, and the length of the LAA in men and women ≥20 years were 1.16, 1.83, and 2.59 cm, and 1.07, 1.66, and 2.53 cm, respectively. The +1 standard deviation values for the mean diameter of the orifice, width of the body, and the length of the LAA in men and women ≥20 years were 1.8, 3.2, and 4.5 cm, and 1.8, 3.2, and 4.1 cm, respectively. In another study of 144 patients with nonvalvular AF, Beinart et al. analyzed the size of the LAA by MRI and angiography as a predictor of stroke and transient ischemic attack (TIA). In the group with stroke and TIA, the mean length and the long and short axis of the neck of the LAA were found to be significantly larger at 3.76, 3.12, and 2.08 cm, respectively, compared to patients without stroke and TIA.75 In our review, we found 3 reported cases in TTE of LAAA with dimensions smaller than the mean dimension reported in the above series. Also, the shape of the LAA is also highly variable with predominantly 4 morphologies described in the literature that includes Chicken Wing, Cactus, Windsock, and Cauliflower types.75 Because of the lack of standard defining criteria of LAAA, we believe that the reporting diagnosis and management of LAAA is fairly arbitrary at best at present. Conventionally, in coronary arteries and aorta, an aneurysm is defined as a 1.5-fold increase in the diameter of a segment of the artery compared to a normal sized segment.76 We propose a similar 5

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size-based criteria for the definition of LAAA. As discussed above, Veinot et al. reported the size of normal LAA in an autopsy series of human hearts.74 Utilizing the +1 standard deviation values in men as the largest normal dimension of the LAA, a LAAA could be defined as a LAA that has dimensions at the diameter of the orifice, width of the body, and the length of the LAA larger than 2.7, 4.8, and 6.75 cm, respectively. A standardized definition of LAAA, we believe, is necessary for better reporting and management of this entity. Conclusion: Aneurysm of left atrial appendage is rare and often an incidental diagnosis during echocardiography. It is important to recognize this entity, as it is associated with cardiovascular morbidity and mortality by predisposing to atrial tachyarrhythmia and thromboembolism. Surgical resection is the standard of treatment in the current literature. Medical management is directed at treatment of thromboembolism and atrial tachyarrhythmia. Acknowledgments: We would like to thank Ronald Polinsky, M.D., Department of Cardiology at Berks Cardiology for his valuable contribution.

References 1. Chowdhury UK, Seth S, Govindappa R, et al: Congenital left atrial appendage aneurysm: a case report and brief review of literature. Heart Lung Circ. 2009;18:412–416. 2. Chen MC, Chang JP, Guo GB, et al: Atrial size reduction as a predictor of the success of radiofrequency maze procedure for chronic atrial fibrillation in patients undergoing concomitant valvular surgery. J Cardiovasc Electrophysiol 2001;12:867–874. 3. Wagshal AB, Applebaum A, Crystal P, et al: Atrial tachycardia as the presenting sign of a left atrial appendage aneurysm. Pacing Clin Electrophysiol 2000;23:283–285. 4. Gold JP, Afifi HY, Ko W, et al: Congential giant aneurysms of the left atrial appendage: diagnosis and management. J Card Surg 1996;11:147–150. 5. Miljak T, Kunze M, Birkemeyer R, et al: First diagnosis of an aneurysm of the left atrial appendage in a 69-year-old female patient. Med Klin (Munich) 2009;104:875–877. 6. Yong HS, Kim EJ, Choi CU: Giant left atrial appendage aneurysm. Eur Heart J 2007;28:2207. 7. Parker JO, Connell WF, Lynn RB: Left atrial aneurysm. Am J Cardiol 1967;20:579–582. 8. Plonska-Gosciniak E, Larysz B, Jurczyk K, et al: Five-chambered heart: A 20-year story of left atrial appendage aneurysm. Eur Heart J 2009;30:1014. 9. Ozmen CA, Alyan O, Altintasß B, et al: Case images: Visualization of a giant left atrial aneurysm by multidetector €rk computed tomography and echocardiography. Tu Kardiyol Dern Arsß 2009;37:76. 10. Sevimli S, Gundogdu F, Aksakal E, et al: A rare congenital anomaly: biatrial appendage aneurysm with atrial and ventricular septal defect. Echocardiography 2007;24:987– 990. 11. Godwin TF, Auger P, Key JA, et al: Intrapericardial aneurysmal dilatation of the left atrial appendage. Circulation 1968;37:397–401. 12. Bramlet DA, Edwards JE: Congenital aneurysm of left atrial appendage. Br Heart J 1981;45:97–100.

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13. Frambach PJ, Geskes GG, Cheriex EC, et al: Giant intrapericardial aneurysm of the left atrial appendage. Eur Heart J 1990;11:848–853. 14. Comess KA, Labate DP, Winter JA, et al: Congenital left atrial appendage aneurysm with intact pericardium: diagnosis by transesophageal echocardiography. Am Heart J 1990;120:992–996. 15. Clarke JR, Zvaigzne CG, Disler D, et al: Giant left atrial appendage aneurysm in a neonate. World J Pediatr Congenit Heart Surg 2012;3:392–395. 16. Hassan M, Said K, El-Hamamsy I, et al: Giant congenital left atrial appendage aneurysm. J Am Coll Cardiol 2013;61:478. 17. Culver DL, Bezante GP, Schwarz KQ, et al: Transesophageal echocardiography in the diagnosis of acquired aneurysms of the left atrial appendage. Clin Cardiol 1993;16:149–151. 18. Cho MJ, Park JA, Lee HD, et al: Congenital left atrial appendage aneurysm diagnosed by fetal echocardiography. J Clin Ultrasound 2010;38:94–96. 19. Zhao J, Ge Y, Yan H, et al: Treatment of congenital aneurysms of the left atrium and left atrial appendage. Tex Heart Inst J 1999;26:136–139. 20. Aytacß A, Oram A, Olga R, et al: Intrapericardial aneurysm of the left atrial appendix. J Cardiovasc Surg (Torino) 1980;21:509–512. 21. Victor S, Nayak VM: Aneurysm of the Left Atrial Appendage. Tex Heart Inst J 2001;28:111–118. 22. Burke RP, Mark JB, Collins JJ Jr, et al: Improved surgical approach to left atrial appendage aneurysm. J Card Surg 1992;7:104–107. 23. Hall J, Dobbs RH: Cerebral emboli from aneurysm of left atrial appendage. Proc R Soc Med 1969;62:911. 24. Parmley LF Jr: Congenital atriomegaly. Circulation 1962;25:553–558. 25. DeSena HC, Niyazov DM, Parrino PE, et al: An unusual cardiac defect in a patient with clinical features overlapping between cardiofaciocutaneous and Noonan syndromes. Congenit Heart Dis 2010;5:70–75. 26. Sloman G, Hunt D, Hare WS: Aneurysmal dilatation of the left atrial appendage. Med J Aust 1969;2:101–102. 27. Williams WG: Dilatation of the left atrial appendage. Br Heart J 1963;25:637–643. 28. Park JS, Lee DH, Han SS, et al: Incidentally found, growing congenital aneurysm of the left atrium. J Korean Med Sci 2003;18:262–266. 29. Munarriz A, Escribano E, Urchaga A, et al: Congenital aneurysm of the left atrial appendage. Eur J Echocardiogr 2008;9:152–154. 30. Gajjar T, Desai N: Giant aneurysm of left atrial appendage–a rare anomaly. Eur J Cardio-Thorac Surg 2011;40:1270. 31. Cujec B, Bharadwaj B, Orchard RC, et al: Transesophageal echocardiography in the diagnosis of left atrial appendage aneurysm. J Am Soc Echocardiogr 1990;3:408–411. 32. Pomerantzeff PMA, Freyre HM, de AlmeidaBrand~ao CM, et al: Aneurysm of the left atrial appendage. Ann Thorac Surg 2002;73:1981–1983. 33. Kawata M, Imanaka K, Matsuoka T, et al: Left atrial appendage aneurysm causes severe mitral regurgitation and heart failure: report of a successfully treated case. J Thorac Cardiovasc Surg 2012;143:e17–e18. 34. Tigen KM, Dogan CE, Guler A, et al: The dangerous fifth chamber: congenital left atrial appendage aneurysm. Cardiovasc J Afr 2012;23:e3–e4. 35. Zhang P-F, Zhang M, Zhang W, et al: Giant aneurysm of the left atrial appendage: detected by real-time 3-dimensional echocardiography. Tex Heart Inst J 2010;37: 129–130. 36. Chockalingam A, Alagesan R, Nandakumar M, et al: Massive left atrial appendage aneurysm presenting as supraventricular tachycardia. Indian Heart J 2003;55:379–381.

Left Atrial Appendage Aneurysm, Systematic Review

37. Ulucam M, Muderrisoglu H, Sezgin A: Giant left atrial appendage aneurysm: the third ventricle!. Int J Cardiovasc Imaging 2005;21:225–230. 38. Wilson D, Kalra N, Brody EA, et al: Left atrial appendage aneurysm-a rare anomaly with an atypical presentation. Congenit Heart Dis 2009;4:489–493. 39. Bhattarai A, Padalino MA, Stellin G: Congenital giant aneurysm of the left atrial appendage in an infant. Cardiol Young 2011;21:697–699. € rk E, Kanadasßi M, Yerdelen VD, et al: Left atrial 40. Acartu appendage aneurysm presenting with recurrent embolic strokes. Int J Cardiovasc Imaging 2003;19: 495–497.  n de la Torre RS: Aneurysm of the left atrial append41. Leo age. Rev Esp Cardiol 2005;58:320. 42. Yaliniz H, Salih OK, Alhan C, et al: Congenital intrapericardial left atrial appendage aneurysm. J Cardiovasc Surg (Torino) 2007;48:109–111. 43. Tanabe T, Ishizaka M, Ohta S, et al: Intrapericardial aneurysm of the left atrial appendage. Thorax 1980;35: 151–153. 44. Hoffmann U, Hamed N, Herold C, et al: Radiological signs of a left atrial aneurysm. Eur Radiol 2000;10: 1332–1334. 45. Sarin SS, Bindra T, Chhabra GS: A giant left atrial appendage aneurysm with a large pinball-like thrombus in a 2 year old. Ann Pediatr Cardiol 2012;5:215–216. 46. Nagai T, Higaki J, Okayama H: Cardiovascular flashlight. Atrial tachycardia in congenital left atrial appendage aneurysm: three-dimensional computed tomography imaging with electro-anatomical mapping. Eur Heart J 2010;31:1590. 47. Kwan CM, Tsai L-M, Lin L-J, et al: Congenital left atrial appendage aneurysm with thrombus formation: Diagnosis by transesophageal echocardiography. J Clin Ultrasound 1993;21:480–483. 48. De Feyter PJ, Zienkowicz BS, Heidendal GA, et al: Radionuclide angiography in the diagnosis of congenital intrapericardial aneurysm of the left atrial appendage. Thorax 1980;35:154–155. 49. Coselli JS, Beall AC Jr, Ziaddi GM: Congenital intrapericardial aneurysmal dilatation of the left atrial appendage. Ann Thorac Surg 1985;39:466–467. € n T, Kahveci G, Gu € ler A, et al: Huge intrapericardial 50. Akgu €rk Kardiyol Dern aneurysm of the left atrial appendage. Tu Arsß 2009;37:212. 51. Ganeshakrishnan KI, Khandeparkar JM, Natrajan VM, et al: Congenital intrapericardial aneurysm of the left-atrial appendage. Thorac Cardiovasc Surg 1992;40:382–384. 52. Kiaii B, Doll N, Kuehl M, et al: Minimal invasive endoscopic resection of a giant left atrial appendage aneurysm. Ann Thorac Surg 2004;77:1437–1438. 53. Mathur A, Zehr KJ, Sinak LJ, et al: Left atrial appendage aneurysm. Ann Thorac Surg 2005;79:1392. 54. LaBarre TR, Stamato NJ, Hwang MH, et al: Left atrial appendage aneurysm with associated anomalous pulmonary venous drainage. Am Heart J 1987;114: 1243–1245. 55. Soleimani A, Sattarzadeh R: Left atrial appendage aneurysm: a rare cause of paroxysmal supraventricular tachycardia. Heart Lung Circ 2008;17:246–247. 56. Krishnamoorthy KM: Aneurysm of the left atrial appendage. Cardiology 2002;97:233–234. 57. Lipkin D, Colli A, Somerville J: Aneurysmal dilatation of left atrial appendage diagnosed by cross sectional echocardiography and surgically removed. Br Heart J 1985;53: 69–71. 58. Smeglin A, Merchan J, Maysky M, et al: Images in cardiovascular medicine: Giant left atrial appendage aneurysm. Circulation 2008;118:2393–2394.

59. Shirazi SH, Fiedotin A: Intrapericardial left atrial appendage aneurysm. Can J Cardiol 1987;3:164–167. 60. Pome G, Pelenghi S, Grassi M, et al: Congenital intrapericardial aneurysm of the left atrial appendage. Ann Thorac Surg 2000;69:1569–1571. 61. Dumitrescu A, Walsh KP, Wood AE: Giant left atrial appendage with a common ventricular-appendicular wall and an abnormal course of the circumflex coronary artery in an asymptomatic 18-month-old girl. Pediatr Cardiol 2008;29:431–433. 62. Bilge M, Yasar AS, Bozkurt M, et al: Left atrial appendage aneurysm secondary to eccentric severe ischemic mitral regurgitation. Echocardiography 2009;26:1225–1227. 63. Krueger SK, Ferlic RM, Mooring PK: Left atrial appendage aneurysm: correlation of noninvasive with clinical and surgical findings: report of a case. Circulation 1975;52: 732–738. 64. Van der Hauwaert LG, Dumoulin M, Daenen W, et al: Aneurysm of the left atrial appendage. Clin Cardiol 1979;2:49–51. €hn A, Schreiber C, Vogt M: Congenital left atrial 65. Ku appendage aneurysm in a 2-year-old boy. Eur Heart J 2006;27:959. 66. Baburaj AK, Rameshwara T, Vellachamy KA, et al: Offpump excision of left atrial appendage aneurysm: a case report. Heart Surg Forum 2006;9:E478–E479. 67. Veiga VC, Rojas SSO, Silva Junior A, et al: Left atrial appendage aneurysm: echocardiographic diagnostic. Arq Bras Cardiol 2008;90:e36–e38. €llmann S, et al: Symptomatic 68. Conradi G, Deetjen A, Mo atrial fibrillation as the first symptom of a giant left atrial appendage aneurysm. Clin Res Cardiol 2006;95:614–616. 69. Atchison FW, Rehfeldt KH: Echo rounds: congenital left atrial appendage aneurysm. Anesth Analg 2011;112: 1303–1305. 70. Shih Y-J, Lin Y-C, Tsai Y-T, et al: Left atrial appendage aneurysm with paroxysmal atrial fibrillation. Heart Surg Forum 2012;15:E1–E3. 71. Lekkerkerker JC, Jaarsma W, Cramer MJM: Congenital giant aneurysm of the left atrial appendage. Heart Br Card Soc 2005;91:e21. 72. Vagefi PA, Choudhry M, Hilgenberg AD: Excision of an aneurysm of the left atrial appendage. J Thorac Cardiovasc Surg 2007;133:822–823. 73. Foale RA, Gibson TC, Guyer DE, et al: Congenital aneurysms of the left atrium: recognition by cross-sectional echocardiography. Circulation 1982;66:1065–1069. 74. Veinot JP, Harrity PJ, Gentile F, et al: Anatomy of the normal left atrial appendage: a quantitative study of agerelated changes in 500 autopsy hearts: implications for echocardiographic examination. Circulation 1997;96: 3112–3115. 75. Beinart R, Heist EK, Newell JB, et al: Left atrial appendage dimensions predict the risk of stroke/TIA in patients with atrial fibrillation. J Cardiovasc Electrophysiol 2011;22:10– 15. 76. Cohen P, OʼGara PT: Coronary artery aneurysms: A review of the natural history, pathophysiology, and management. Cardiol Rev 2008;16:301–304.

Supporting Information Additional Supporting Information may be found in the online version of this article: Movie Clip S1. Mid-esophageal 90° views demonstrating giant left atrial appendage aneurysm. Note the presence of spontaneous echo contrast in the left atrial appendage aneurysm.

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Left atrial appendage aneurysm: a systematic review of 82 cases.

Aneurysm of the left atrial appendage is rare. We sought to systematically review the published literature on left atrial appendage aneurysm (LAAA) to...
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