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• W2074278625 abstract "We report two cases of adults who had occlusion of the left coronary artery ostium by the left coronary cusp. Both patients were treated with the Bentall procedure, and both revealed a good, long-term course. We offer proper strategy of perioperative management consisting of precise preoperative diagnosis with a multimodality imaging study and adequate operative technique. We report two cases of adults who had occlusion of the left coronary artery ostium by the left coronary cusp. Both patients were treated with the Bentall procedure, and both revealed a good, long-term course. We offer proper strategy of perioperative management consisting of precise preoperative diagnosis with a multimodality imaging study and adequate operative technique. Occlusion of the left coronary artery ostium by the left coronary cusp is a rare and potentially fatal anomaly because it may cause sudden death. There are only a small number of reported cases and long-term prognosis for these younger patients who had surgical treatment are unknown [1Danie J.J. Verheught A.P. Supravalvular aortic stenosis.Circulation. 1958; 18: 902-908Crossref PubMed Scopus (42) Google Scholar, 2Kurosawa H. Wagenaar S.S. Becker A.E. Sudden death in a youth A case report of quadricuspid aortic valve with isolation of origin of left coronary artery.Br Heart J. 1981; 46: 211-215Crossref PubMed Scopus (48) Google Scholar, 3Kalimi R. Palazzo R.S. Graver L.M. Occlusion of left coronary artery ostium by an aortic valve cusp.Ann Thorac Surg. 2000; 69: 637-639Abstract Full Text Full Text PDF PubMed Scopus (20) Google Scholar]. A systematic, perioperative strategy is needed for safe and durable treatment for this disorder. A 61-year-old woman was referred to Shizuoka City Hospital because of occlusion of the left coronary ostium. She had a history of depression and had no family history of sudden death or cardiac disorder. Two years before she had been referred to us, she had cardiac work-up for a systolic murmur at another hospital. She was diagnosed with an occlusion of the left coronary artery (LCA) ostium and aortic stenosis. Although surgical treatment was recommended, the patient refused the operation because she had been asymptomatic. In November 2009, she first experienced chest pain. After she took nitroglycerine prescribed by a home doctor, she fell into shock and was transferred to another hospital. She was successfully resuscitated with norepinephrine administration. After this episode, she accepted an operation and was referred to our hospital. Her physical examination showed a systolic murmur of Levine III/VI maximal at the right sternal border. She had a normal mental status and no facial characteristics suggestive of Williams syndrome. Her electrocardiography at rest was normal, and a thallium scintigraphy revealed myocardial ischemia in the entire LCA territory. Her transthoracic echocardiography showed significant aortic stenosis. In a coronary angiography, the LCA orifice could not be cannulated. The right coronary artery was normal. The LCA with normal anatomy was retrogradely filled from the right coronary artery followed by contrast pooling in the left Valsalva sinus with the blind sac. A proximal portion of the left anterior descending coronary arterial (LAD) branch seemed to be stenotic (Fig 1). A contrast-enhanced computed tomographic scan revealed a more dilated ascending aorta (48-mm diameter), only slight narrowing at the sinotubular junction, and a membrane-like structure in the left Valsalva sinus (Fig 2). We planned the Bentall procedure, replacement of the ascending aorta, and coronary bypass grafting to the LAD with the left internal thoracic artery. The operation was performed through a median sternotomy. Extracorporeal circulation was established with bi-caval venous cannulation and ascending aortic and femoral arterial cannulation. Core cooling was started. Cardioplegia was retrogradely introduced after the aorta was cross clamped. The ascending aorta was opened. The left coronary cusp (LCC) totally obliterated the LCA ostium. After excising the LCC, a normal opening of the LCA was found. There was no supravalvular ridge. The bicuspitized aortic valve was stenotic (Fig 3). After resection of the LCC, cardioplegia could be injected either antegradely or retrogradely. Aortic open distal anastomosis was accomplished under hypothermic circulatory arrest and continuous retrograde cerebral perfusion. The Bentall procedure was completed with a 25-mm SJM valve (St. Jude Medical, St. Paul, MN) and a 28-mm Dacron graft (J Graft NEO [Japan Lifeline, Shinagawa, Tokyo, Japan]). Both coronary arteries were reattached as the Carrel patch. Finally, the left internal thoracic artery was anastomosed to the LAD. The histopathologic examination of the aortic wall and the left coronary cusp demonstrated almost a normal amount of elastic fiber and aortic medial structure. The patient's postoperative recovery was uneventful, and she is doing well 9 months after the operation.Fig 2(A) Contrast-enhanced computed tomographic (CT) scan showing aortic root structure. See membrane-like septum isolating the left coronary artery (LCA) ostium. (B) A CT reconstructed aortic valve image showing bicuspid morphology. The LCA (black asterisk) and fused LCA (white arrow).View Large Image Figure ViewerDownload (PPT)Fig 3An intraoperative surgeon's view of patient 1 showing the left coronary cusp (asterisk). Noncoronary and right coronary cusps were held by forceps. No supravalvular ridge was found.View Large Image Figure ViewerDownload (PPT) A 37-year-old man had been worked-up for chest pain, which was diagnosed as supravalvular aortic stenosis (SVAS), valvular aortic stenosis, and occlusion of the LCA by the LCC in 1997. He had no other anomaly. Intraoperatively, an apparent supravalvular ridge and tiny hole in the occluding LCC were found (Fig 4). The Bentall procedure was carried out with interposing graft for LCA re-implantation. The patient has been doing well 13 years after the operation. Occlusion or isolation of coronary artery ostium was first reported by Danie and Verheught [1Danie J.J. Verheught A.P. Supravalvular aortic stenosis.Circulation. 1958; 18: 902-908Crossref PubMed Scopus (42) Google Scholar] in 1958. This disorder is so rare that approximately only 15 cases have ever been reported since then [2Kurosawa H. Wagenaar S.S. Becker A.E. Sudden death in a youth A case report of quadricuspid aortic valve with isolation of origin of left coronary artery.Br Heart J. 1981; 46: 211-215Crossref PubMed Scopus (48) Google Scholar, 3Kalimi R. Palazzo R.S. Graver L.M. Occlusion of left coronary artery ostium by an aortic valve cusp.Ann Thorac Surg. 2000; 69: 637-639Abstract Full Text Full Text PDF PubMed Scopus (20) Google Scholar], and we add 2 cases here. Most authors consistently stated that this anomaly would be life threatening. In some patients, the first event may be sudden death, and in other patients, symptoms such as cardiac murmur, chest pain, or shortness of breath may lead to cardiac work-up for a cure. The severity of this disorder is influenced by many factors as follows: age, richness of intercoronary collateral channel, presence of communication of occluding cusp, degree of myocardial ischemia, involvement of right or left coronary ostium, function of aortic valve, and presence of supravalvular stenosis. Pathologically, this anomaly is believed to be congenital and related to SVAS, as noted “forme fruste of SVAS” by Kurosawa and colleagues [2Kurosawa H. Wagenaar S.S. Becker A.E. Sudden death in a youth A case report of quadricuspid aortic valve with isolation of origin of left coronary artery.Br Heart J. 1981; 46: 211-215Crossref PubMed Scopus (48) Google Scholar]. However, as with our first case, the cases without SVAS were also reported. Supravalvular aortic stenosis is known to be related to the elastin gene mutation [4Kumar A. Olson T.M. Thibodeau S.N. et al.Confirmation of linkage of supravalvular aortic stenosis to the elastin gene on chromosome 7q.Am J Cardiol. 1994; 74: 1281-1283Abstract Full Text PDF PubMed Scopus (13) Google Scholar], but no reports showed relation of the gene and the development of this anomaly. We cannot perform molecular biological assay, but resected tissue samples of this case were preserved for it in the future. More remains to be analyzed regarding the pathophysiology in this rare disorder. This anomaly could be clearly distinct with single coronary, left main atresia, or obstruction of left main associated with SVAS [5Thistlethwaite P.A. Madani M.M. Kriett J.M. Mihoan K. Jamieson S.W. Surgical management of congenital obstruction of the left main coronary artery with supravalvular aortic stenosis.J Thorac Cardiovasc Surg. 2000; 120: 1040-1046Abstract Full Text Full Text PDF PubMed Scopus (43) Google Scholar] from imaging studies. Ectopic origin of LCA ostium from the left ventricle is an extremely rare condition in which only two cases had been reported [6Pirelli L. Yu P.J. Srichai M.B. Khvilivitzky K. Angelini P. Grau J.B. Ectopic origin of left coronary ostium from left ventricle, with occlusive membrane.Tex Heart Inst J. 2008; 35: 162-165PubMed Google Scholar, 7Okuyama M. Kubota I. Miura T. Nagamine S. Yokoyama K. Anomalous origin of the right coronary artery from the left ventricle in an adult.Jpn Heart J. 1995; 36: 115-118Crossref PubMed Scopus (9) Google Scholar]. This can also be differentiated in our patients undoubtedly by computed tomography and angiogram. In the point of surgical view, we should discuss approximately 2 points: (1) myocardial protection and (2) procedure selection. Poor surgical outcomes in very early cases may reflect the importance of adequate intraoperative myocardial protection for this condition. We used retrograde and antegrade cardioplegia injection, and in the first patient the left internal thoracic artery was bypassed in fear of myocardial infarction in the LAD area. In most cases, aortic valve replacement is preferred, and the Bentall procedure was done in only one patient [8Washiyama N. Kazui T. Takinami M. et al.Aortic regurgitation with dilatation of ascending aorta and right coronary artery occlusion by a rudimentary aortic cusp.Ann Thorac Surg. 2001; 72: 919-921Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar]. We selected the Bentall procedure for both patients, because it is superior in full resection of the pathologic root tissue, and safe and reliable coronary reconstruction. Our second patient has been followed-up for 13 years postoperatively, longer than any other reported cases. In conclusion, we had two adult cases of occlusion of LCA ostium by fusion of the LCC with satisfactory results. The Bentall procedure will be the operation of choice for this rare anomaly." @default.
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• W2074278625 title "Occlusion of Left Coronary Artery Ostium by Left Coronary Cusp Fusion" @default.
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