Ann Thorac Surg 2005;80:1912-1914
© 2005 The Society of Thoracic Surgeons
Case report
Idiopathic Inflammatory Aneurysm of the Ascending Aorta
Hrvoje Gasparovic, MD
a
,
Luigino Nascimben, MD
b
,
David W. Kindelberger, MD
c
,
John G. Byrne, MD
a
,
*
a Division of Cardiac Surgery, Boston, Massachusetts, USA
b Department of Anesthesiology, Brigham and Women's Hospital, Boston, Massachusetts, USA
c Department of Pathology, Brigham and Women's Hospital, Boston, Massachusetts, USA
Accepted for publication June 11, 2004.
* Address correspondence to Dr Byrne, Division of Cardiac Surgery, Brigham and Women's Hospital, 75 Francis St, Boston, MA02115 (Email: jbyrne{at}partners.org).
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Abstract
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Inflammatory aneurysms of the ascending aorta are exceedingly rare. The principal cause of ascending aortic aneurysms remains arteriosclerosis. We present a patient with an ascending aortic aneurysm caused by active inflammatory disease superimposed on complex atherosclerotic disease of the aorta.
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Introduction
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Inflammatory ascending aortic aneurysms are exceedingly rare with only six cases reported in the literature to date. Inflammatory aortic aneurysms have been hypothesized to occur due to an excessive autoimmune response to components of atherosclerotic plaque [1]. Genetic factors and tobacco use have also been implicated in the pathogenesis of this condition [2]. The indications for surgical intervention include the absolute size of the aneurysm as well as its progression, the concomitant development of aortic regurgitation, or dissection.
A 72-year-old woman presented with progressive shortness of breath and chest pain. Her past medical history was remarkable for partial nephrectomy for renal cell carcinoma. She also had multiple risk factors for accelerated atherosclerotic disease, including dyslipidemia and a 25 pack-per-year smoking history. On routine follow-up abdominal computed tomographic scan for her history of renal cell carcinoma, an enlarged proximal abdominal aorta was noted. Further diagnostic evaluation included contrast enhanced computed tomography of her chest, which indicated that she had a 4.9 cm ascending aortic aneurysm (Fig 1)A. A suspicion was raised that an intramural hematoma was present along with her recent history of chest pain; therefore we decided it was urgent to proceed with surgery. The patient denied any history of temporal headache, visual anomalies, jaw claudication, or proximal muscle weakness, which could have alerted a suspicion for an autoimmune origin of the aneurysm. Because an inflammatory cause of the aneurysm was not preoperatively suspected, no specific markers of inflammation were drawn. Preoperative coronary angiography demonstrated a left anterior descending coronary artery lesion. The aorta and the valvular apparatus were imaged intraoperatively using transesophageal echocardiography (Fig 1B). No evidence of aortic valve insufficiency was found. At surgery, cardiopulmonary bypass was established, the patient was cooled to 20°C, and the left internal mammary artery was placed to the left anterior descending coronary artery. The inspection of the ascending aorta showed that its size was not the sole abnormality. Its appearance was polished white and fibrotic, indicating that inflammation played a significant component in the presenting pathology (Fig 2). The ascending aorta was then resected, and the sinotubular junction, coronary ostia, and aortic valve were scrutinized. We found no deviation in the position of the coronary ostia. The aortic root was of normal diameter and the valve showed no structural anomaly. The anastomosis between the proximal aorta and a 32-mm Hemashield graft (Boston Scientific, Boston, MA) was then fashioned. The distal portion of the ascending aorta was resected and the distal anastomosis was constructed using an "open" technique because the aneurysm extended close to the innominate artery. Circulatory arrest, myocardial ischemia, and cardiopulmonary bypass durations were 15, 86, and 135 minutes, respectively. Histopathological analysis of the aortic specimen revealed severe active aortitis. The media was found to be profoundly enlarged with violent medial degeneration, necrosis, and scarring. This was accompanied by adventitial fibrosis and lymphoplasmocytic infiltration (Fig 3). Moderate complex arteriosclerosis was evident in the specimen. No distinctive giant cells were seen. The patient's postoperative course was complicated by an embolic cerebrovascular accident, which resulted in minimal left arm weakness. She also had a tachycardia–bradycardia syndrome develop that was managed by the placement of a permanent pacemaker. She was discharged from the hospital on postoperative day 14.
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Fig 1. (A) Contrast enhanced computed tomographic image of the ascending aortic aneurysm with a suspected intramural hematoma. (B) Intraoperative transesophageal echocardiogram demonstrating the ascending aortic aneurysm. Appropriate aortic valve leaflet coaptation is also visualized.
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Fig 2. Intraoperative view of the glistening white wall of the ascending aortic aneurysm. (AO = aorta; IV = innominate vein; RA = right atrium.)
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Fig 3. (A) The resected wall of the ascending aortic aneurysm shows severe circumferential thickening. (B) Hematoxylin & eosin stain (x20) reveals a dense mononuclear inflammatory infiltrate involving the aortic adventitia with dense adventitial fibrosis.
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Comment
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Inflammatory ascending aortic aneurysms are exceedingly rare [3, 4]. Connery and colleagues [1] were the first to report this condition. A thorough review of the literature revealed a total of six reported cases [1, 3–7]. Most inflammatory aortic aneurysms have a topographic predilection for the infrarenal aorta, which is the case for approximately 3% to 10% of the case reports [2]. The descending thoracic aorta has also been reported as a site for inflammatory aneurysms, although its involvement is much less frequent. The principal event is the presentation of an unknown antigen on the aortic wall followed by infiltration of macrophages T and B lymphocytes. The result is an increase in the production of cytokines and proteolytic enzymes. The aftermath of this upsurge in pro-inflammatory activity is an accelerated turnover of the aortic wall proteins, elastin, and collagen [2]. The cause of inflammatory diseases of the aorta is multifactorial. Hereditary, environmental factors, and infectious factors all play an important role. The role of viral agents in the development of inflammatory aortic aneurysms has recently been postulated [8]. The patient presented here had an ascending aortic aneurysm develop secondary to severe active aortitis complicated by medial necrosis. Her preoperative evaluation suggested that the ascending aortic aneurysm was complicated by the formation of an intramural hematoma. This suspicion was not confirmed intraoperatively. The failure of computed tomographic angiography and transesophageal echocardiography to differentiate between an inflammatory ascending aortic aneurysm and an intramural aortic hemorrhage has already been identified in the literature [3, 5]. The circumferential solidification of the aortic wall in our patient was of inflammatory origin, which was confirmed by histologic analysis. The infrequent and sporadic reports on inflammatory ascending aortic aneurysms prevent us from gathering conclusive evidence regarding their natural history. Therefore a strategy of regular computed tomographic follow-up scans should be embraced to compensate for the anecdotal experience with this clinical entity.
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References
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- Connery CP, Descalzi ME, Kirschner R. Inflammatory aneurysm of the ascending aortaan unreported entity. J Cardiovasc Surg 1993;35:33-34.
- Rasmussen TE, Hallett Jr JW. Inflammatory aortic aneurysmsA clinical review with new perspectives in pathogenesis. Ann Surg 1997;225:155-164.[Medline]
- Kunzli A, von Segesser LK, Vogt PR, et al. Inflammatory aneurysm of the ascending aorta Ann Thorac Surg 1998;65:1132-1133.[Abstract/Free Full Text]
- Girardi LN, Coselli JS. Inflammatory aneurysm of the ascending aorta and aortic arch Ann Thorac Surg 1997;64:251-253.[Abstract/Free Full Text]
- Federmann M, Attenhofer CH, Jenni R. Inflammatory aneurysm of the ascending aorta mimicking intramural hemorrhage Circulation 1996;93:1477-1478.
- Roth M, Schonburg M, Klovekorn WP, Bauer EP. Inflammatory aneurysm of the ascending aorta Eur J Cardiothorac Surg 2001;19:214.[Free Full Text]
- Roth M, Lemke P, Bohle RM, Klovekorn WP, Bauer EP. Inflammatory aneurysm of the ascending thoracic aorta J Thorac Cardiovasc Surg 2002;123:822-824.[Free Full Text]
- Tanaka S, Komori K, Okadome K, Sugimachi K, Mori R. Detection of active cytomegalovirus infection in inflammatory aortic aneurysms with RNA polymerase chain reaction J Vasc Surg 1994;20:235-243.[Medline]
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Abstract
Introduction
