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Atrial Septal Aneurysm and Stroke – A Report of Two Cases LAURA POANTĂ, D.L. DUMITRAŞCU, DANIELA FODOR, ADRIANA ALBU “I. Haţieganu” University of Medicine and Pharmacy, Department of Internal Medicine, Cluj-Napoca, Romania Atrial septal aneurysm (ASA) is an uncommon lesion which often associates other cardiac abnormalities such as patent foramen ovale or mitral valve prolapse. More than half of the cases with ASA associate an interatrial shunt which can explain cerebral embolism and consecutive ischemic stroke. Case summary. We present two cases which were admitted to our hospital with minor complaints. Personal histories excluded cardiac disease, high blood pressure, smoking, and included ischemic stroke in both cases. Physical examination revealed no abnormalities. Electrocardiogram showed right bundle branch block in one case, lab findings were in normal limits. Cardiac ultrasound (transthoracic and transesophageal) revealed in both cases an ASA, one with bidirectional equidistant excursion without visible interatrial shunt, and one with unilateral excursion and a small shunt. Conclusion. The ASA diagnosis should be remembered as an alternative cause in a patient with a history of stroke, or with developing stroke, and without risk factors for cerebrovascular disease. Key words: atrial septal aneurysm, stroke. Atrial septal aneurysm (ASA) is an uncommon lesion with a varying prevalence in studies with transthoracic echocardiography (TTE), transesophagian echocardiography (TEE) and autopsies [1][2]. The prevalence of ASA varies with the examination method: transthoracic echocardiographic studies estimate the rate to be between 0.08% and 1.2%, in autopsies the prevalence reported was 1%, and in more recent studies using TEE the prevalence was between 2% and 10% [1][3][4]. It often associates other cardiac abnormalities such as patent foramen ovale, atrial septal defects, ventricular septal defects, valvular prolapse, patent ductus arteriosus, Ebstein’s anomaly, and tricuspid and pulmonary atresia, as well as acquired heart diseases including valvular disease, cardiomyopathy, systemic and pulmonary hypertension, ischemic heart disease, arrhythmias and thrombus formation [1][2][4]. More than half of the cases with ASA associate an interatrial shunt which can explain a consecutive ischemic stroke. Atrial septal aneurysm has been considered a potential cardiac source of embolism in recent years (transient ischemic attacks and cerebrovascular accidents), but the role of atrial septal aneurysm as a risk factor is not very well defined [2–4]. In a multicentric study, the authors concluded that atrial septal aneurysm could be considered a risk factor for cardiogenic embolism in a significant subgroup ROM. J. INTERN. MED., 2008, 46, 4, 357–360 of patients [5]. The report from the Stroke Prevention: Assessment of Risk in a Community (SPARC) study demonstrated that atrial septal aneurysm is associated with an increased risk of stroke. This study established the prevalence of ASA in the general population at 2.2%. The true prevalence of ASA was underestimated before the routine use of echocardiography, especially TEE. It seems that paradoxical embolism is the predominant mechanism of cardioembolism in ASA, according to SPARC [6]. In contrast, a prospective long-term study suggested that the risk of cerebrovascular events is low in a patient population with incidental atrial septal aneurysm [7]. Atrial septal aneurysm was defined as a bulging over 15 mm beyond the plane of the atrial septum as measured by transoesophageal echocardiography. Atrial septal aneurysm was classified according to Hanley’s diagnostic criteria, modified by Pearson to include type 1C [8]. The type of aneurysm was determined according to morphology and bulging in four or five types [8][9]. CASE ONE MI, fifty years old, female, was admitted in our hospital with dyspepsia. Her personal history excluded ischemic heart disease, high blood pressure, and smoking. Laura Poantă et al. 358 She had an ischemic stroke seven years ago which was CT diagnosed at the time. Physical examination revealed no abnormalities. Blood pressure was 120/70 mmHg in supine position, on admission in the hospital. Rest electrocardiogram and lab findings were in normal limits. The patient had normal carotid arteries on Doppler ultrasound. 2 Cardiac ultrasound found an atrial septal aneurysm, confirmed by TEE (ASA type 5 according to [9]), with a base width of 2.2 cm, with bidirectional and equidistant excursion throughout the cardiac cycle, without interatrial shunt or intra atrial thrombi (Fig. 1). Fig. 1. – Apical four chambers view, atrial septal aneurysm (arrow). CASE TWO DE, 55 years old, female, was admitted with osteoarthritis. Her personal history also excluded ischemic heart disease, high blood pressure, and smoking; she also had an ischemic stroke three years ago, CT confirmed and completely recovered. Physical examination revealed no abnormalities. Blood pressure was 135/78 mmHg in supine position on admission. Rest electrocardiogram showed right bundle brunch block. Laboratory findings were in normal limits and she had normal carotid arteries. Cardiac transthoracic ultrasound found an atrial septal aneurysm, confirmed by TEE (ASA type 1 R, according to [9]), with base width 1.8 cm, with unidirectional excursion and interatrial left-toright shunt, without intra atrial thrombi, and with normal heart chambers (Fig. 2). The following parameters were also evaluated: length of the atrial septal aneurysm, maximal protrusion of the atrial septal aneurysm beyond the plane of the atrial septum and the direction of the maximal protrusion, oscillation of the atrial septal aneurysm during a normal respiratory cycle, thickening of the atrial septal aneurysm. DISCUSSION We choose to present those two cases because this diagnosis is uncommon, although the incidence of atrial septal aneurysm in the normal population is still controversial. Many reasons explain this: different diagnostic criteria, methodology used, and age of patients and lack of recognition [1][3]. We cannot state for sure that there is a definite connection between stroke and ASA in those two cases, but the probability is high. The embolic mechanism cannot be excluded in the first case, despite the fact that the shunt is missing, as contrast TEE is in fact more sensitive than TEE color Doppler 3 359 Atrial septal aneurysm and stroke Fig. 2. – Transthoracic view, atrial septal aneurysm (arrow). alone in detecting interatrial shunt of small dimensions [9]. So we cannot rule out the paradox embolism in one case; other possible causes of cardioembolic events are: thrombus formation inside ASA, undiagnosed transient atrial arrhythmias, small or rapidly resolving thrombi in ASA. Both cases received medical treatment. Many authors observed that a number of patients with atrial septal aneurysm presented cerebral ischemic events, which were otherwise unexplained, as in our both cases [1][3][5]. A study of Nighoghossian et al. found atrial septal aneurysm in the 34.5% of the 79 patients who had an unexplained stroke [10]. In subjects less than 55 years of age, a lower prevalence of atherosclerosis makes a diagnosis of “cryptogenic” stroke (stroke without other clear cause) more frequent than in older subjects. Atrial septal aneurysm has been described recently as the only potential source of embolism in a significant proportion of subjects who had sustained a transient ischemic attack [11]. Mattioli et al. have also demonstrated that patients who have sustained cryptogenic stroke, and have atrial septal aneurysm, constitute a subgroup which is at a higher risk of recurrent stroke [1]. CONCLUSION The atrial septal aneurysm diagnosis should be remembered as an alternative cause in patients with a history of stroke, or with developing stroke, and without risk factors for cerebrovascular disease, especially under fifty five years old. Anevrismul de sept interatrial (ASI) este o leziune mai puţin frecventă care se asociază adesea cu alte anomalii cardiace cum ar fi foramen ovale patent sau prolaps de valvă mitrală. Mai mult de jumătate dintre cazurile de ASI au asociat un şunt interatrial care ar putea explica accidentele ischemice cerebrale consecutive emboliei. Prezentare de caz. Raportăm două cazuri internate în clinica noastră cu simptome minore. Antecedentele personale patologice exclud bolile cardiace, Laura Poantă et al. 360 4 hipertensiunea arterială, fumatul, şi constau în accidente vasculare cerebrale ischemice în ambele situaţii. Examenul obiectiv a fost în limite normale, electrocardiograma arată bloc major de ramură dreaptă într-un caz, fiind normală în celălalt, iar analizele de laborator au fost normale. Ecocardiografia (transtoracică şi transesofagiană) a relevat în ambele cazuri un ASI, unul cu excursie bidirecţională, echidistantă, fără şunt vizibil, celălalt cu excursie unidirecţională şi un şunt mic. Concluzie. Diagnosticul de ASI trebuie avut în vedere ca şi o posibilă cauză de accident vascular cerebral ischemic la un pacient cu antecedente personale patologice pozitive sau cu debut recent şi fără alţi factori de risc detectabili pentru boala cerebrovasculară. Corresponding author: Laura Poantă 2–4, Clinicilor Str., 400006, Cluj-Napoca, Romania, Mobile phone: 0040744894190 E-mail address: [email protected] REFERENCES 1. MATTIOLI A.V., AQUILINA M., OLDANI A., LONGHINI C., MATTIOLI G., Atrial septal aneurysm as a cardioembolic source in adult patients with stroke and normal carotid arteries. Eur. Heart J., 2001; 22: 261–268. 2. FEIGENBAUM H., Echocardiography. 6th ed. Philadelphia: Lippincott Williams & Wilkins; 2005. p. 93, 187–191. 3. 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HANLEY P.C., TAJIK A.J., HYNES J.K. et al., Diagnosis and classification of atrial septal aneurysm by two-dimensional echocardiography: report of 80 consecutive cases. J. Am. Coll. Cardiol., 1985; 6:1370–1382. 9. OLIVARES-REYES A. et al., Atrial Septal Aneurysm: A new classification in 205 adults. J. Am. Soc. Echocardiogr., 1997; 10: 644–56. 10. NIGHOGHOSSIAN N., PERINETTI M., BARTHELET M., ADELEINE P., TROUILLAS P., Potential cardioembolic sources of stroke in patients less than 60 years of age. Eur. Heart J., 1996; 17: 590–4. 11. BEACOCK D.J., WATT V.B., OAKLEY G.D., MOHAMMAD A., Paradoxical embolism with a patent foramen ovale and atrial septal aneurysm. Eur. J. Echocardiogr., 2006; 7 (2): 171–174. Received October 13, 2008