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Transcatheter occlusion of the ruptured sinus of Valsalva aneurysm with an Amplatzer duct occluder
International Journal of Cardiology 129 (2008) 81 – 85 www.elsevier.com/locate/ijcard
Transcatheter occlusion of the ruptured sinus of Valsalva aneurysm with an Amplatzer duct occluder Shi-Hua Zhao a,⁎, Chao-Wu Yan a , Xian-Yang Zhu b , Jian-Jun Li a,⁎, Nai-Xun Xu c , Shi-Linag Jiang a , Zhong-Ying Xu a , Cheng Wang a , Wen-Hui Wu a , Hu Hai-Bo a , Shi-Guo Li a , Zan-Kai Ye a , Hao Wang a a
Department of Radiology and Cardiology, Cardiovascular Institute and Fuwai Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100037, China b Department of Cardiology, PLA General Hospital of Shengyang, Shengyan 110000, China c Department of Radiology, Tianjing Chest Hospital, Tianjing 300000, China Received 16 February 2007; received in revised form 21 May 2007; accepted 15 June 2007 Available online 30 July 2007
Abstract Background: Ruptured sinus of Valsalva aneurysm (RSVA) can be associated with ventricular septal defects or isolated lesions. Percutaneous transcatheter closure of RSVA has been an alternative strategy to surgery. The results of transcatheter closure of the RSVA in 10 patients were presented. Methods: From January 2000 to May 2006, 10 patients (4 males, 6 females) aged from 7 years to 69 years (mean ages 37 ± 18.8 years) were involved in the present report. The diagnosis of RSVA was made based on a combination of several imaging modalities. Of them, 9 patients were identified as congenital cause and one did as acquired RSVA. Two-dimensional and color Doppler echocardiography revealed the rupture of right coronary sinus into right ventricle in 5 cases and into right atrium in 3 cases, while non-coronary sinus ruptured into right atrium in 2 cases. Aortogram showed that the estimated size of the defect was 6.2 ± 2.3 mm (2–10 mm). After the establishment of the arteriovenous wire loop, Amplatzer duct occluder (ADO) was deployed by antegrade venous approach in all patients. Results: ADO with 1–3mm larger than the defect was used. All defects were successfully occluded without any complications. On the follow-up, echocardiography showed neither residual shunt nor aortic regurgitation, and there was also no device embolization, infective endocarditis in any of the patients. Conclusions: Transcatheter closure is a feasible and effective alternative for both congenital and acquired RSVA. However, long-term followup is mandatory. © 2007 Elsevier Ireland Ltd. All rights reserved. Keywords: Congenital heart disease; Aneurysm; Amplatzer duct occluder; Transcatheter
1. Introduction Though the sinus of Valsalva aneurysm occurs rarely in the western world, the incidence in Asian populations is higher [1]. The congenital deficiency of elastic and muscular
⁎ Corresponding authors. Zhao is to be contacted at Tel.: +86 10 88398404. Li, Tel.: +86 10 88396077. E-mail addresses: [email protected] (S.-H. Zhao), [email protected] (J.-J. Li). 0167-5273/$ - see front matter © 2007 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijcard.2007.06.022
tissue in the aortic sinus leads to the formation of aneurysm. Most of them originate from the right coronary sinus, followed by the non-coronary sinus, and rarely from the left coronary sinus. In addition, infection, operation, trauma, degenerative and inflammatory processes can result in acquired sinus of Valsalva aneurysm, which usually affects more than one sinus of Valsalva. Patients are often asymptomatic before the rupture of the sinus of Valsalva aneurysm (RSVA), however, the worsening of symptoms commonly occurs when the sinus of Valsalva aneurysm ruptures, which usually occurs in adolescence and adulthood.
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duct occluder for transcatheter closure including one case with acquired RSVA. 2. Materials and methods 2.1. Patients From January 2000 to May 2006, 10 patients (4 male, 6 females) were accepted for transcatheter closure of RSVA, and their ages were from 7 to 69 years (mean ages 37 ± 18.8 years). Of them, 9 patients were due to the rupture of congenital sinus of Valsalva aneurysm. Another did after his surgical repair of ventricular septal defect (VSD). Dyspnea upon physical exertion was found in 8 patients (among them, 2 patients also presented left heart failure). No symptomatic presentation did in two other patients, and their defects were identified by murmur and echocardiography followed by detailed evaluation included electrocardiograms, chest
Fig. 1. A 7-year-old boy, the aneurysm of right Valsalva sinus was found after the surgical repair of VSD. In the long axial oblique view, aortagram showed formation of aneurysm of right Valsalva sinus rupturing into right ventricle. The opening of aneurysm was about 2 mm (A). Repeated aortagram after the deployment of 6/4 mm ADO. The shunt disappeared and no aortic insufficiency was found (B).
Surgical repair has become the traditional methods to treat the patients with RSVA since 1957, and the mortality is low with good long-term results. But some problems limit the application of surgical repair, for instance, hemodynamic instability increase the risk of operation, and the recurrence of the lesion or aortic regurgitation requires the second sternotomy. Therefore, it is necessary to develop an alternative method to treat RSVA without sternotomy. Since Cullen's report in 1994, transcatheter closure of RSVA had developed and several kinds of occlusion devices had been applied. However, all previous reports except one were case report [4], and devices used in those study were entirely not same in different cases. [2–7] In addition, no acquired RSVA was attempted for transcatheter closure. In this study, we present ten cases with RSVA using Amplatzer
Fig. 2. The aneurysm of non coronary sinus ruptured into right atrium. In the right anterior oblique view, the opening of aneurysm was about 4 mm (A). Repeated aortagram after the deployment of 8/6 mm ADO (B).
S.-H. Zhao et al. / International Journal of Cardiology 129 (2008) 81–85
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Fig 3. The aneurysm of right coronary sinus ruptured into right atrium, which was showed by aortagram in the long axial oblique view (A), and by transthoracic echocardiography in parasternal aortic and left atrial short axis view (B). The opening of aneurysm was about 6 mm, and mild aortic insufficiency occurred. After the deployment of 10/8 mm ADO, repeated aortagram (C) and transoesophageal echocardiography (D) were performed. The shunt disappeared completely without the worsening of aortic insufficiency. Arrow, the opening of RSVA; Arrowhead, ADO.
radiographs, and echocardiography. Sinus tachycardias were demonstrated in six patients by the electrocardiogram. Marked enlargement of the heart and increased pulmonary arterial blood pressure was found in 5 patients, mild enlargement in 3, and normal sized hearts did in 2 patients. Echocardiography confirmed the left to right shunt from the right coronary sinus to right ventricle in 5 patients, from the right coronary sinus to right atrium in 3 patients, and from non coronary sinus to right atrium in 2 patients. Associated cardiac lesions included: mild aortic insufficiency in 4 patients; mild mitral insufficiency did in 1 patient. One patient had a VSD that was operated on several month ago. All 10 patients underwent attempted transcatheter closure of the RSVA after obtaining informed consent from the patients and/or parents. 2.2. Device design Amplatzer duct occluder (ADO, AGA Medical Corporation, Golden Valley, MN) was used for all patients in the
present study. The device design has been described in detail according the previous reports [8]. 2.3. Procedure Under local anesthesia, the femoral arterial and venous introducing sheaths were inserted into right femoral artery and vein percutaneously. All patients underwent routine right and left heart catherterization and a complete haemodynamic data were obtained in each cardiac chamber and great arteries. Then, a 5F pigtail catheter was introduced into left ventricle and aorta. In the long axial oblique view, the left ventriculogram was taken to rule out VSD. Then, in the long axial oblique view and right anterior oblique view, the aortogram was carried out to delineate the origin, the rupture sites, the narrowest opening and the anatomy of RSVA (Figs. 1A, 2A, 3A). The aortic and mitral insufficiency was also assessed. To eliminate the possibility of the lesion of right coronary artery (RCA), selective right coronary
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S.-H. Zhao et al. / International Journal of Cardiology 129 (2008) 81–85
Table 1 The clinical data of ruptured sinus of Valsalva aneurysm Patient No
Sex
Age (years)
Ruptured sinus of Valsalva aneurysm
Size of the opening (mm)
Size of ADO
Qp/Qs
PAP[S/D(M)]
Association
Residual shunt
1 2 3 4 5 6 7 8 9 10 n = 10
M F F F F M F M M F 4M6F
7 54 30 57 69 32 31 34 16 40 37 [18.8]
RCS (into RV) RCS (into RV) RCS (into RV) RCS (into RV) NCS (into RA) RCS (into RA) RCS (into RA) RCS (into RA) NCS (into RA) RCS (into RV) 8RCS 2NCS
2 7 7 10 4 8 8 6 5 5 6.2 [2.3]
4 8 8 12 6 10 10 8 8 8 8.2 [2.2]
1.2 2.0 1.4 2.7 1.7 2.6 2.4 1.19 1.5 1.9 1.86 [0.56]
20/4(11) 28/9(15) 34/22(24) 42/26(28) 32/16(22) 40/11(21) 47/10(22) 28/10(18) 46/20(28) 35/20(23) S 35.2 [8.6] M 21.2 [5.4]
VSD a AI (mild) MI (mild) AI (mild) AI (mild) no no AI (mild) no no
No No Small b⁎ No No No No No No No
a
Resulting from the surgical repair of ventricular septal defect. UCG showed that the shunt disappeared next day; RCS = right coronary sinus; NCS = non coronary sinus; RV, = right ventricle; RA = right atrium; VSD = ventricular septal defect; AI = aortic insufficiency; MI = mitral insufficiency; PAP = pulmonary arterial pressure (S, systolic; D, diastolic; M, mean); Size of ADO = the narrowest segment of occluder (the pulmonary segment of ADO). b
arteriography was carried out, especially in older patients. Based on the measurement of the aortogram, the proper ADO was selected, and the diameter of pulmonary segment of ADO was larger 1 to 3 mm than the narrowest diameter of the openings of aneurysm. Next, an arterial-venous wire loop was established from right femoral artery to right femoral vein via the RSVA. Under the guidance of fluoroscopy and echocardiography, the ADO was introduced from the venous route, and was deployed in the opening of the ruptured sinus of Valsalva aneurysm. 10 min later, aortogram was repeated to assess the occlusion of the ruptured sinus of Valsalva aneurysm and to eliminate the possibility of deterioration of aortic insufficiency resulting from the occluders (Figs. 1B, 2B, 3C). When the aneurysm originated from the right coronary sinus, selective right coronary arteriography was repeated to avoid the ostium of the RCA being occluded by occluder. The ADO were released finally if above findings were satisfactory. In the procedure, transthoracic or transoesophageal echocardiography was performed to assess the RSVA (Fig. 3B) and guide the deployment of ADO (Fig. 3D). Prophylactic antibiotics were routinely administrated after the procedure for three days. All patients returned home after 2-day observation in the heart ward. For each patient, chest X-ray, electrocardiogram and echocardiography were performed at one day, one month, and serially at 3 to 6 month intervals. 3. Results 10 cases were involved in this study, and the detailed clinical data was showed in Table 1. 5 patients had rupture sites of right coronary sinus aneurysm into right ventricle, and 3 patients into right atrium. The non coronary sinus aneurysm ruptured into right atrium in another 2 patients. The size of the opening measured by aortogram was from 2 mm to 10 mm. The Qp/Qs was 1.19 to 2.7. The pulmonary
arterial pressures of three patients were within normal limit, and those of the other seven were mild elevated. Successful occlusion of the RSVA was achieved in all ten patients. The procedure time was 56 to 82 min and the fluoroscopic time was 27 to 48 min. Trace residual shunt was found in one patient and it disappeared the next day, which was confirmed by echocardiography. No complication was found in the procedure. On the follow-up (3 months), the symptoms of these four patients disappeared, and there was no device embolization, infective endocarditis, and aortic regurgitation. 4. Discussion The relative defect of elastic fibers and muscular tissue leads to the formation of aneurysm of the sinus of Valsalva, which progresses over time and usually rupture into one chamber finally. Because the defect occurs above the aortic annulus, the aortic valves usually escaped the encroachment of aneurysm in the beginning. The incidence of the right coronary sinus rupturing into the right ventricle or atrium was highest, and there was usually a distance between the aneurysms and the opening of right coronary artery, which makes it possible to close the RSVA percutaneously. The ruptured aneurysms of non coronary sinus occurred secondly, and it usually ruptured into right atrium, which makes it easy and safe for transcatheter closure of defect. The Rashkind umbrella was the first device that was used to close the RSVA [2]. Then the Gianturco coil and ADO were applied to occlude RSVA [3–7]. In addition, there was at least one case of RSVA in which was treated using AGA septal occluder [4]. Arterial route was needed for the deployment of Rashkind umbrella and Gianturco coil, and this may potentially cause arterial damage for the large sheath, so the application of these two devices was limited. With the application of ADO in RSVA, the procedure became more convenient and safe for the device was delivered from the
S.-H. Zhao et al. / International Journal of Cardiology 129 (2008) 81–85
femoral vein after the establishment of arterio-venous loop. Transcatheter closure of two sites of RSVA was also attempted [5]. Because the maximum size of ADO is 16/14 mm and this is too small for large RSVA, Amplatzer septal occluder was attempted in some research, but the result was unsatisfying [4]. In this study, ADO was used to close RSVA percutaneously. All origins of RSVA were from right coronary cusp and all ruptured into right ventricle in the present study. In patients with RSVA, most originate from the right coronary sinus and ruptured into the right ventricle or right atrium, especially in Asian, and they were the target populations for transcatheter closure of RSVA. This makes it possible for transcatheter closure of most RSVA without anomalies requiring surgical management. In these patients, the 7-year-old boy was a special case. In his age of 3 years, echocardiography found a peri-membraneous VSD (5⁎5 mm) without anomalies of aorta, and surgical repair was carried out elsewhere. Though the shunt from LV to RV disappeared, the shunt from the right coronary sinus to right ventricle was found in the post-operation echocardiography. Because no RSVA was found in the preoperation echocardiography and surgical detection, we thought that the RSVA resulted from the “post-surgical complication”. The size of opening measured by aortogram was 2 mm (Fig. 1A) and the left ventriculogram ruled out the shunt in the ventricular level. To avoid the second sternotomy, transcatheter closure of RSVA was attempted and a 6/4 mm ADO was deployed in the site of RSVA. To our knowledge, this is the first reported case that applies ADO to close the acquired RSVA. Because RSVA were usually associated with VSD or aortic insufficiency, left ventriculogram and aortogram were required to rule out these associations before transcatheter closure of RSVA. The deployment of ADO has the potential to interfere with the cusps of aortic valves and the opening of right coronary artery, so the repeat aortogram and selective right coronary arteriography were necessary, especially for RSVA of non coronary sinus. No complications were found in this study. In the present, the technique was limited in several heart centers on a small
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scale, and the associated experiences were relevant absent. Only one report about the complications can be found: the obstruction of right ventricular outlet tract and persistent hemolysis occurred after transcatheter closure of RSVA, and the patient was finally referred for surgical repair [4]. The immediate and short to moderate results were good, and the longest follow-up lasted 2 to 96 months[4]. Though large progression had been made for transcatheter closure of RSVA, many problems still needed to be solved, such as indication, contra-indication, the selection of occlusion device and so on. 5. Conclusions Not only congenital but also acquired RSVA can be closed effectively with ADO based on a catheter approach, which should become an alternative of surgical repair. The long-term follow-up is, however, mandatory. References [1] Chu Shu-hsum, Hung Chi-Ren, How Sou-Sien, et al. Ruptured aneurysm of the sinus of Valsalva in oriental patients. J Thorac Cardiovasc Surg 1990;99:288–92. [2] Cullen S, Somerville J, Redington A. Transcatheter closure of a ruptured aneurysm of the sinus of Valsalva. Br Heart J 1994;71:479–80. [3] Rao PS, Bromberg BI, Jureidini SB, Fiore AC. Transcatheter occlusion of ruptured sinus of valsalva aneurysm: innovative use of available technology. Catheter Cardiovasc Interv 2003;58:130–4. [4] Arora R, Trehan V, Rangasetty UM, Mukhopadhyay S, Thakur AK, Kalra GS. Transcatheter closure of ruptured sinus of Valsalva aneurysm. J Interv Cardiol 2004;17:53–8. [5] Fedson S, Jolly N, Lang RM, Hijazi ZM. Percutaneous closure of a ruptured sinus of Valsalva aneurysm using the Amplatzer duct occluder. Catheter Cardiovasc Interv 2003;58:406–11. [6] Hijazi ZM. Ruptured sinus of Valsalva aneurysm: management options. Catheter Cardiovasc Interv 2003;58:135–6. [7] Abidin N, Clarke B, Khattar RS. Percutaneous closure of ruptured sinus of Valsalva aneurysm using an Amplatzer occluder device. Heart 2005;91:244. [8] Masura J, Walsh KP, Thanopoulous B, et al. Catheter closure of moderate- to large-sized patent ductus arteriosus using the new Amplatzer duct occluder: immediate and short-term results. J Am Coll Cardiol 1998;31:878–82.
Transcatheter occlusion of the ruptured sinus of Valsalva aneurysm with an Amplatzer duct occluder Shi-Hua Zhao a,⁎, Chao-Wu Yan a , Xian-Yang Zhu b , Jian-Jun Li a,⁎, Nai-Xun Xu c , Shi-Linag Jiang a , Zhong-Ying Xu a , Cheng Wang a , Wen-Hui Wu a , Hu Hai-Bo a , Shi-Guo Li a , Zan-Kai Ye a , Hao Wang a a
Department of Radiology and Cardiology, Cardiovascular Institute and Fuwai Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100037, China b Department of Cardiology, PLA General Hospital of Shengyang, Shengyan 110000, China c Department of Radiology, Tianjing Chest Hospital, Tianjing 300000, China Received 16 February 2007; received in revised form 21 May 2007; accepted 15 June 2007 Available online 30 July 2007
Abstract Background: Ruptured sinus of Valsalva aneurysm (RSVA) can be associated with ventricular septal defects or isolated lesions. Percutaneous transcatheter closure of RSVA has been an alternative strategy to surgery. The results of transcatheter closure of the RSVA in 10 patients were presented. Methods: From January 2000 to May 2006, 10 patients (4 males, 6 females) aged from 7 years to 69 years (mean ages 37 ± 18.8 years) were involved in the present report. The diagnosis of RSVA was made based on a combination of several imaging modalities. Of them, 9 patients were identified as congenital cause and one did as acquired RSVA. Two-dimensional and color Doppler echocardiography revealed the rupture of right coronary sinus into right ventricle in 5 cases and into right atrium in 3 cases, while non-coronary sinus ruptured into right atrium in 2 cases. Aortogram showed that the estimated size of the defect was 6.2 ± 2.3 mm (2–10 mm). After the establishment of the arteriovenous wire loop, Amplatzer duct occluder (ADO) was deployed by antegrade venous approach in all patients. Results: ADO with 1–3mm larger than the defect was used. All defects were successfully occluded without any complications. On the follow-up, echocardiography showed neither residual shunt nor aortic regurgitation, and there was also no device embolization, infective endocarditis in any of the patients. Conclusions: Transcatheter closure is a feasible and effective alternative for both congenital and acquired RSVA. However, long-term followup is mandatory. © 2007 Elsevier Ireland Ltd. All rights reserved. Keywords: Congenital heart disease; Aneurysm; Amplatzer duct occluder; Transcatheter
1. Introduction Though the sinus of Valsalva aneurysm occurs rarely in the western world, the incidence in Asian populations is higher [1]. The congenital deficiency of elastic and muscular
⁎ Corresponding authors. Zhao is to be contacted at Tel.: +86 10 88398404. Li, Tel.: +86 10 88396077. E-mail addresses: [email protected] (S.-H. Zhao), [email protected] (J.-J. Li). 0167-5273/$ - see front matter © 2007 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijcard.2007.06.022
tissue in the aortic sinus leads to the formation of aneurysm. Most of them originate from the right coronary sinus, followed by the non-coronary sinus, and rarely from the left coronary sinus. In addition, infection, operation, trauma, degenerative and inflammatory processes can result in acquired sinus of Valsalva aneurysm, which usually affects more than one sinus of Valsalva. Patients are often asymptomatic before the rupture of the sinus of Valsalva aneurysm (RSVA), however, the worsening of symptoms commonly occurs when the sinus of Valsalva aneurysm ruptures, which usually occurs in adolescence and adulthood.
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S.-H. Zhao et al. / International Journal of Cardiology 129 (2008) 81–85
duct occluder for transcatheter closure including one case with acquired RSVA. 2. Materials and methods 2.1. Patients From January 2000 to May 2006, 10 patients (4 male, 6 females) were accepted for transcatheter closure of RSVA, and their ages were from 7 to 69 years (mean ages 37 ± 18.8 years). Of them, 9 patients were due to the rupture of congenital sinus of Valsalva aneurysm. Another did after his surgical repair of ventricular septal defect (VSD). Dyspnea upon physical exertion was found in 8 patients (among them, 2 patients also presented left heart failure). No symptomatic presentation did in two other patients, and their defects were identified by murmur and echocardiography followed by detailed evaluation included electrocardiograms, chest
Fig. 1. A 7-year-old boy, the aneurysm of right Valsalva sinus was found after the surgical repair of VSD. In the long axial oblique view, aortagram showed formation of aneurysm of right Valsalva sinus rupturing into right ventricle. The opening of aneurysm was about 2 mm (A). Repeated aortagram after the deployment of 6/4 mm ADO. The shunt disappeared and no aortic insufficiency was found (B).
Surgical repair has become the traditional methods to treat the patients with RSVA since 1957, and the mortality is low with good long-term results. But some problems limit the application of surgical repair, for instance, hemodynamic instability increase the risk of operation, and the recurrence of the lesion or aortic regurgitation requires the second sternotomy. Therefore, it is necessary to develop an alternative method to treat RSVA without sternotomy. Since Cullen's report in 1994, transcatheter closure of RSVA had developed and several kinds of occlusion devices had been applied. However, all previous reports except one were case report [4], and devices used in those study were entirely not same in different cases. [2–7] In addition, no acquired RSVA was attempted for transcatheter closure. In this study, we present ten cases with RSVA using Amplatzer
Fig. 2. The aneurysm of non coronary sinus ruptured into right atrium. In the right anterior oblique view, the opening of aneurysm was about 4 mm (A). Repeated aortagram after the deployment of 8/6 mm ADO (B).
S.-H. Zhao et al. / International Journal of Cardiology 129 (2008) 81–85
83
Fig 3. The aneurysm of right coronary sinus ruptured into right atrium, which was showed by aortagram in the long axial oblique view (A), and by transthoracic echocardiography in parasternal aortic and left atrial short axis view (B). The opening of aneurysm was about 6 mm, and mild aortic insufficiency occurred. After the deployment of 10/8 mm ADO, repeated aortagram (C) and transoesophageal echocardiography (D) were performed. The shunt disappeared completely without the worsening of aortic insufficiency. Arrow, the opening of RSVA; Arrowhead, ADO.
radiographs, and echocardiography. Sinus tachycardias were demonstrated in six patients by the electrocardiogram. Marked enlargement of the heart and increased pulmonary arterial blood pressure was found in 5 patients, mild enlargement in 3, and normal sized hearts did in 2 patients. Echocardiography confirmed the left to right shunt from the right coronary sinus to right ventricle in 5 patients, from the right coronary sinus to right atrium in 3 patients, and from non coronary sinus to right atrium in 2 patients. Associated cardiac lesions included: mild aortic insufficiency in 4 patients; mild mitral insufficiency did in 1 patient. One patient had a VSD that was operated on several month ago. All 10 patients underwent attempted transcatheter closure of the RSVA after obtaining informed consent from the patients and/or parents. 2.2. Device design Amplatzer duct occluder (ADO, AGA Medical Corporation, Golden Valley, MN) was used for all patients in the
present study. The device design has been described in detail according the previous reports [8]. 2.3. Procedure Under local anesthesia, the femoral arterial and venous introducing sheaths were inserted into right femoral artery and vein percutaneously. All patients underwent routine right and left heart catherterization and a complete haemodynamic data were obtained in each cardiac chamber and great arteries. Then, a 5F pigtail catheter was introduced into left ventricle and aorta. In the long axial oblique view, the left ventriculogram was taken to rule out VSD. Then, in the long axial oblique view and right anterior oblique view, the aortogram was carried out to delineate the origin, the rupture sites, the narrowest opening and the anatomy of RSVA (Figs. 1A, 2A, 3A). The aortic and mitral insufficiency was also assessed. To eliminate the possibility of the lesion of right coronary artery (RCA), selective right coronary
84
S.-H. Zhao et al. / International Journal of Cardiology 129 (2008) 81–85
Table 1 The clinical data of ruptured sinus of Valsalva aneurysm Patient No
Sex
Age (years)
Ruptured sinus of Valsalva aneurysm
Size of the opening (mm)
Size of ADO
Qp/Qs
PAP[S/D(M)]
Association
Residual shunt
1 2 3 4 5 6 7 8 9 10 n = 10
M F F F F M F M M F 4M6F
7 54 30 57 69 32 31 34 16 40 37 [18.8]
RCS (into RV) RCS (into RV) RCS (into RV) RCS (into RV) NCS (into RA) RCS (into RA) RCS (into RA) RCS (into RA) NCS (into RA) RCS (into RV) 8RCS 2NCS
2 7 7 10 4 8 8 6 5 5 6.2 [2.3]
4 8 8 12 6 10 10 8 8 8 8.2 [2.2]
1.2 2.0 1.4 2.7 1.7 2.6 2.4 1.19 1.5 1.9 1.86 [0.56]
20/4(11) 28/9(15) 34/22(24) 42/26(28) 32/16(22) 40/11(21) 47/10(22) 28/10(18) 46/20(28) 35/20(23) S 35.2 [8.6] M 21.2 [5.4]
VSD a AI (mild) MI (mild) AI (mild) AI (mild) no no AI (mild) no no
No No Small b⁎ No No No No No No No
a
Resulting from the surgical repair of ventricular septal defect. UCG showed that the shunt disappeared next day; RCS = right coronary sinus; NCS = non coronary sinus; RV, = right ventricle; RA = right atrium; VSD = ventricular septal defect; AI = aortic insufficiency; MI = mitral insufficiency; PAP = pulmonary arterial pressure (S, systolic; D, diastolic; M, mean); Size of ADO = the narrowest segment of occluder (the pulmonary segment of ADO). b
arteriography was carried out, especially in older patients. Based on the measurement of the aortogram, the proper ADO was selected, and the diameter of pulmonary segment of ADO was larger 1 to 3 mm than the narrowest diameter of the openings of aneurysm. Next, an arterial-venous wire loop was established from right femoral artery to right femoral vein via the RSVA. Under the guidance of fluoroscopy and echocardiography, the ADO was introduced from the venous route, and was deployed in the opening of the ruptured sinus of Valsalva aneurysm. 10 min later, aortogram was repeated to assess the occlusion of the ruptured sinus of Valsalva aneurysm and to eliminate the possibility of deterioration of aortic insufficiency resulting from the occluders (Figs. 1B, 2B, 3C). When the aneurysm originated from the right coronary sinus, selective right coronary arteriography was repeated to avoid the ostium of the RCA being occluded by occluder. The ADO were released finally if above findings were satisfactory. In the procedure, transthoracic or transoesophageal echocardiography was performed to assess the RSVA (Fig. 3B) and guide the deployment of ADO (Fig. 3D). Prophylactic antibiotics were routinely administrated after the procedure for three days. All patients returned home after 2-day observation in the heart ward. For each patient, chest X-ray, electrocardiogram and echocardiography were performed at one day, one month, and serially at 3 to 6 month intervals. 3. Results 10 cases were involved in this study, and the detailed clinical data was showed in Table 1. 5 patients had rupture sites of right coronary sinus aneurysm into right ventricle, and 3 patients into right atrium. The non coronary sinus aneurysm ruptured into right atrium in another 2 patients. The size of the opening measured by aortogram was from 2 mm to 10 mm. The Qp/Qs was 1.19 to 2.7. The pulmonary
arterial pressures of three patients were within normal limit, and those of the other seven were mild elevated. Successful occlusion of the RSVA was achieved in all ten patients. The procedure time was 56 to 82 min and the fluoroscopic time was 27 to 48 min. Trace residual shunt was found in one patient and it disappeared the next day, which was confirmed by echocardiography. No complication was found in the procedure. On the follow-up (3 months), the symptoms of these four patients disappeared, and there was no device embolization, infective endocarditis, and aortic regurgitation. 4. Discussion The relative defect of elastic fibers and muscular tissue leads to the formation of aneurysm of the sinus of Valsalva, which progresses over time and usually rupture into one chamber finally. Because the defect occurs above the aortic annulus, the aortic valves usually escaped the encroachment of aneurysm in the beginning. The incidence of the right coronary sinus rupturing into the right ventricle or atrium was highest, and there was usually a distance between the aneurysms and the opening of right coronary artery, which makes it possible to close the RSVA percutaneously. The ruptured aneurysms of non coronary sinus occurred secondly, and it usually ruptured into right atrium, which makes it easy and safe for transcatheter closure of defect. The Rashkind umbrella was the first device that was used to close the RSVA [2]. Then the Gianturco coil and ADO were applied to occlude RSVA [3–7]. In addition, there was at least one case of RSVA in which was treated using AGA septal occluder [4]. Arterial route was needed for the deployment of Rashkind umbrella and Gianturco coil, and this may potentially cause arterial damage for the large sheath, so the application of these two devices was limited. With the application of ADO in RSVA, the procedure became more convenient and safe for the device was delivered from the
S.-H. Zhao et al. / International Journal of Cardiology 129 (2008) 81–85
femoral vein after the establishment of arterio-venous loop. Transcatheter closure of two sites of RSVA was also attempted [5]. Because the maximum size of ADO is 16/14 mm and this is too small for large RSVA, Amplatzer septal occluder was attempted in some research, but the result was unsatisfying [4]. In this study, ADO was used to close RSVA percutaneously. All origins of RSVA were from right coronary cusp and all ruptured into right ventricle in the present study. In patients with RSVA, most originate from the right coronary sinus and ruptured into the right ventricle or right atrium, especially in Asian, and they were the target populations for transcatheter closure of RSVA. This makes it possible for transcatheter closure of most RSVA without anomalies requiring surgical management. In these patients, the 7-year-old boy was a special case. In his age of 3 years, echocardiography found a peri-membraneous VSD (5⁎5 mm) without anomalies of aorta, and surgical repair was carried out elsewhere. Though the shunt from LV to RV disappeared, the shunt from the right coronary sinus to right ventricle was found in the post-operation echocardiography. Because no RSVA was found in the preoperation echocardiography and surgical detection, we thought that the RSVA resulted from the “post-surgical complication”. The size of opening measured by aortogram was 2 mm (Fig. 1A) and the left ventriculogram ruled out the shunt in the ventricular level. To avoid the second sternotomy, transcatheter closure of RSVA was attempted and a 6/4 mm ADO was deployed in the site of RSVA. To our knowledge, this is the first reported case that applies ADO to close the acquired RSVA. Because RSVA were usually associated with VSD or aortic insufficiency, left ventriculogram and aortogram were required to rule out these associations before transcatheter closure of RSVA. The deployment of ADO has the potential to interfere with the cusps of aortic valves and the opening of right coronary artery, so the repeat aortogram and selective right coronary arteriography were necessary, especially for RSVA of non coronary sinus. No complications were found in this study. In the present, the technique was limited in several heart centers on a small
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scale, and the associated experiences were relevant absent. Only one report about the complications can be found: the obstruction of right ventricular outlet tract and persistent hemolysis occurred after transcatheter closure of RSVA, and the patient was finally referred for surgical repair [4]. The immediate and short to moderate results were good, and the longest follow-up lasted 2 to 96 months[4]. Though large progression had been made for transcatheter closure of RSVA, many problems still needed to be solved, such as indication, contra-indication, the selection of occlusion device and so on. 5. Conclusions Not only congenital but also acquired RSVA can be closed effectively with ADO based on a catheter approach, which should become an alternative of surgical repair. The long-term follow-up is, however, mandatory. References [1] Chu Shu-hsum, Hung Chi-Ren, How Sou-Sien, et al. Ruptured aneurysm of the sinus of Valsalva in oriental patients. J Thorac Cardiovasc Surg 1990;99:288–92. [2] Cullen S, Somerville J, Redington A. Transcatheter closure of a ruptured aneurysm of the sinus of Valsalva. Br Heart J 1994;71:479–80. [3] Rao PS, Bromberg BI, Jureidini SB, Fiore AC. Transcatheter occlusion of ruptured sinus of valsalva aneurysm: innovative use of available technology. Catheter Cardiovasc Interv 2003;58:130–4. [4] Arora R, Trehan V, Rangasetty UM, Mukhopadhyay S, Thakur AK, Kalra GS. Transcatheter closure of ruptured sinus of Valsalva aneurysm. J Interv Cardiol 2004;17:53–8. [5] Fedson S, Jolly N, Lang RM, Hijazi ZM. Percutaneous closure of a ruptured sinus of Valsalva aneurysm using the Amplatzer duct occluder. Catheter Cardiovasc Interv 2003;58:406–11. [6] Hijazi ZM. Ruptured sinus of Valsalva aneurysm: management options. Catheter Cardiovasc Interv 2003;58:135–6. [7] Abidin N, Clarke B, Khattar RS. Percutaneous closure of ruptured sinus of Valsalva aneurysm using an Amplatzer occluder device. Heart 2005;91:244. [8] Masura J, Walsh KP, Thanopoulous B, et al. Catheter closure of moderate- to large-sized patent ductus arteriosus using the new Amplatzer duct occluder: immediate and short-term results. J Am Coll Cardiol 1998;31:878–82.