Ann Thorac Surg 2009;88:994-996. doi:10.1016/j.athoracsur.2009.01.033
© 2009 The Society of Thoracic Surgeons
Case Reports
An Atypical Cause of Aortic Valve Prolapse
Yuki Okamoto, MD*,
Masahiko Matsumoto, MD,
Hidenori Inoue, MD
Second Department of Surgery, Faculty of Medicine, University of Yamanashi, Yamanashi, Japan
Accepted for publication January 13, 2009.
* Address correspondence to Dr Okamoto, 1110 Shimokato, Chuo city, Yamanashi, 409-3898, Japan (Email: yamanashimedical{at}yahoo.co.jp).
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Abstract
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We encountered a surgical case of aortic valve prolapse caused by a fissure formed in the commissural region and coronary cusp, and we achieved a favorable outcome. On pathologic examination, the fissured valve was found to be myxoid degeneration with ruptured elastic fibers and clustering of foamy macrophages, and it was diagnosed as an atherosclerotic change not associated with rheumatic change. The fissure appeared to develop because of hypertensive stress in a region rendered vulnerable by atherosclerosis. Although this mechanism is atypical, it should be recognized that poorly controlled hypertension can cause aortic valve prolapse and induce acute heart failure.
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Introduction
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Aortic valve prolapse, not associated with infective endocarditis, aortic dissection, bicuspid aortic valve, or trauma, is very rare [1–5]. We report the extremely rare case of a patient with aortic regurgitation caused by a fissure formed between the cusp and commissural region.
A 68-year-old man with a history of hypertension without medication was admitted to our hospital for examination and treatment of sudden onset dyspnea. He had no history of trauma, chest blow, and valve disease previously. On initial examination, his blood pressure was systolic hypertension (180/66 mm Hg). He was in sinus tachycardia at 110 beats per minutes, and he was noted to have a diastolic regurgitant murmur. His white blood cell count was 7,980/µL and C-reactive protein 7.75 mg/dL, indicating elevation of inflammatory reaction. His blood, urine, and phlegm culture tests and a serologic test for syphilis were negative. Chest roentgenograms showed the features of pulmonary congestion. Transthoracic echocardiography showed severe aortic regurgitation without vegetation and ventricular septal defect, and the left ventricular ejection fraction was 65%. Moreover, transesophageal echocardiography demonstrated the diastolic prolapse of noncoronary cusp without aortic dissection and aortic annular dilatation (Fig 1). Contrast enhanced computed tomography revealed no evidence of aortic dissection. Coronary angiography was performed, but there were no findings of coronary artery stenosis. Cardiac catheterization indicated mild pulmonary hypertension with 34 mm Hg of pulmonary artery wedge pressure and 45 mm Hg of mean pulmonary artery pressure. Urgent operation was planned because medical control of his heart failure could not go well.
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Fig 1. Transesophageal echocardiography demonstrating prolapse of noncoronary cusp (arrow) without aortic dissection.
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At surgery, the aortic valve was tricuspid and almost intact (Fig 2A). No annular dilatation and no aortic dissection were noted. There was no calcification of the aortic valve; however, a fissure formed between the cusp and commissural region and extended to the annulus was found (Fig 2B). There was no evidence of vegetation around the aortic valve and no findings of typical infective endocarditis. Fissured annulus was repaired with autologous pericardium and then the aortic valve was replaced in the supra-annular position using a 21-mm Carpentier-Edwards pericardial valve (Edwards Lifesciences, Irvine, CA). The postoperative course was uneventful; he was doing well without any cardiac events at 9 months after surgery. No infection or thrombus was noted on pathologic examination. Outgrowth of vitrified connective tissue, myxoid degeneration, ruptured elastic fibers, and clustering of foamy macrophages were noted in the destroyed cusps and annulus; these changes were consistent with atherosclerosis (Fig 3).
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Fig 2. Intraoperative picture of (A) tricuspid aortic valve without aortic dissection and vegetation, and (B) a fissure between the cusp and commissural region (white arrow). The fissure extended to the annulus (black arrow). (LCC = left coronary cusp; NCC = noncoronary cusp; RCC = right coronary cusp.)
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Fig 3. (A) Pathology of the noncoronary cusp (arrowhead) and fissured commissural region (arrow). (Hematoxylin & eosin stain; x40.) (B) Histologic picture of fissured commissural region shows the characteristic morphologic features of an atherosclerosis. (Hematoxylin & eosin stain; x200.)
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Comment
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Aortic regurgitation is often caused by infective endocarditis, aortic dissection or trauma, but rare cases due to rupture of the commissural supportive tissue and separation of the commissural region from the aortic wall have also been reported [1–4]. Newcomb and Rowland [2] described aortic valve prolapse associated with localized dehiscence of the aortic wall, and Fukui and colleagues [3] reported aortic valve prolapse associated with local dehiscence of the commissural region without the history of hypertension. However, destruction of the aortic annulus was not found in these two cases. Mok and colleague [4] reported a case associated with atheromatous plaque that caused separation of the commissural region from the aortic wall. Pathologically, the developmental mechanism may have been similar to that in our case, but to our knowledge, there has been no report of a case of aortic valve prolapse in which a fissure formed between the cusp and commissural region and extended to the annulus. The patient had no medical history of trauma, rheumatic fever, aortic dissection, or infective endocarditis, and the aortic valve appeared normal macroscopically. However, atherosclerotic changes were noted in the fissured region on pathologic examination in this case. Lakier and colleagues [5] reported regressive myxoid changes of the aortic valve due to long-term hypertensive stress as a cause for aortic regurgitation based on pathology. An atherosclerotic lesion may have developed during a similar process in our patient, and persistent hypertension may have produced severe stress that led to formation of a fissure in the vulnerable region.
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References
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- Nakajima M, Tsuchiya K, Naito Y, Hibino N, Inoue H. Aortic regurgitation caused by rupture of a well-balanced fibrous strand suspending a degenerative tricuspid aortic valve J Thorac Cardiovasc Surg 2002;124:843-844.[Free Full Text]
- Newcomb AE, Rowland MA. Nontraumatic localized dehiscence of the proximal ascending aorta through an aortic valve commissure Ann Thorac Surg 2004;78:321-323.[Abstract/Free Full Text]
- Fukui T, Shimokawa T, Fumimoto KU, Mananbe S, Ozawa N, Takanashi S. Dehiscence of aortic valve commissure complicated by aortic regurgitation J Thorac Cardiovasc Surg 2008;135:1178-1179.[Free Full Text]
- Mok CK, Tso JW, Khin MA. Dehiscence of an atheromatous plaque at an aortic valve commissure: an unusual cause of acute aortic regurgitation Br Heart J 1988;59:513-515.[Abstract/Free Full Text]
- Lakier JB, Copans H, Rosman HS, et al. Idiopathic degeneration of the aortic valve: a common cause of isolated aortic regurgitation J Am Coll Cardiol 1985;5:347-357.[Medline]
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Abstract
Introduction
