Eur J Cardiothorac Surg 2006;30:806-807
© 2006 Elsevier Science NL
Inflammatory aneurysm of the ascending aorta
Jayesh Dhareshwara,
Anthony L. Estreraa,*,
Michael H. Covinskyb,
Hazim J. Safia
a Department of Cardiothoracic and Vascular Surgery, The University of Texas Health Science Center at Houston Medical School, Memorial Hermann Hospital, 6410 Fannin Street, Suite 450, Houston, TX 77030, United States
b Department of Pathology, The University of Texas Health Science Center at Houston Medical School, Memorial Hermann Hospital, United States
Received 5 April 2006;
received in revised form 22 June 2006;
accepted 6 July 2006.
* Corresponding author. Tel.: +1 713 500 5420; fax: +1 713 500 0656. (Email: Anthony.l.estrera{at}uth.tmc.edu).
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Abstract
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Inflammatory aneurysms of the aorta are usually seen in the infrarenal abdominal aorta and very rarely in the ascending aorta. We present the case of a 76-year-old male with inflammatory aneurysm of the ascending aorta.
Key Words: Inflammatory • Aneurysm • Ascending aorta
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Introduction
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Walker and colleagues [1] first described ‘inflammatory aneurysm’ as a ‘discrete entity with significant differences from atherosclerotic aneurysms’. These aneurysms are usually seen in the infrarenal abdominal aorta, and their presence in the thoracic aorta is extremely uncommon [2,3]. Connery and colleagues [4] were the first to describe an inflammatory aneurysm in the ascending aorta. Crawford and colleagues [5] described five inflammatory aneurysms of the descending thoracic aorta, all in association with a similar aneurysm in the infrarenal position. Typical histologic examination of an ‘inflammatory aneurysm’ shows signs of chronic inflammation in the adventitia with a marked lymphoplasmacytic cell infiltrate and granulation or fibrous tissue proliferation. We report a case of successful repair of an inflammatory aneurysm of the ascending aorta.
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1. Clinical summary
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A 76-year-old Caucasian male was evaluated for cardiovascular and genetic workup after his 52-year-old son was operated upon for acute type A aortic dissection. His past medical history was significant for hypertension and hyperlipidemia. He was previously operated upon for idiopathic retroperitoneal fibrosis. Transthoracic echocardiogram showed a dilated ascending aorta of 5.3 cm. Computed tomographic (CT) scan of the chest confirmed an aneurysm of the ascending aorta and transverse arch with maximal dimension of approximately 5 cm, and no evidence of dissection. Preoperative cardiac evaluation did not reveal any significant coronary artery disease. Surgical intervention was planned because of the 5 cm aortic aneurysm and family history of acute aortic dissection.
Intraoperative transesophageal echocardiography (TEE) showed mild aortic insufficiency and confirmed the absence of dissection. The chest was entered through a median sternotomy. Cardiopulmonary bypass was established by ascending aortic and bicaval cannulation, and myocardial protection was achieved by continuous retrograde and intermittent antegrade cold blood cardioplegia. Cerebral protection was achieved by systemic profound hypothermia to 17.1 °C and retrograde cerebral perfusion. The aneurysm was excised and the ascending aorta and proximal transverse aortic arch were replaced using a 30 mm woven Dacron tube graft with a side arm (total circulatory arrest time 19 min). Aortic valve resuspension was performed to alleviate the mild insufficiency. After complete rewarming, the patient was separated from cardiopulmonary bypass (total bypass time 176 min). The patient was extubated on postoperative day 1, and the remainder of the postoperative course was uneventful.
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2. Discussion
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Inflammatory aortic aneurysms are rare, comprising only 5–10% of all infrarenal aortic aneurysms [1,2]. Involvement of the thoracic aorta is even more uncommon. A thorough review of the English literature revealed only seven case reports of an inflammatory aneurysm of the ascending aorta to date [3,4,6–9]. Crawford and colleagues [5] described a series of 30 patients with inflammatory aneurysms of the abdominal aorta, five of which had involvement of the descending thoracic component. Connery and colleagues [4] were probably the first to describe an inflammatory aneurysm in the ascending aorta.
The majority of the inflammatory aneurysms are symptomatic when first detected clinically. However, this patient did not have any specific symptoms attributable to the aneurysm. He was screened only after his son developed an acute type A dissection of the aorta. Preoperative diagnosis of an inflammatory aneurysm is often difficult. Radiologic criteria for diagnosis include a thickened aortic wall, periaortic fibrosis and contrast enhancement of the aortic wall. None of these features were seen on the preoperative CT scan of this patient. Transesophageal echocardiography may incorrectly identify inflammatory aneurysm as a dissection or intramural hematoma, though it did not in this case. However, suspicion was raised during surgery because of the periaortic adhesions and the thickened calcified aneurysmal wall (Fig. 1
). Grossly, the aortic wall was thickened up to 0.9 cm and showed multiple foci of atherosclerotic plaque. Histopathology confirmed inflammatory aneurysm, showing the typical thickened adventitia with infiltration of the adventitia and media with clusters of plasma cells and lymphocytes (Fig. 2
). An elastic stain (Verhoeff-Van Gieson) highlighted the destruction of the elastic fibers with nearly transmural loss of elastic fibers near one end of the tissue. Giant cells were not identified. Bacterial and fungal cultures obtained from the aneurysm wall were negative. The differential diagnoses for these findings include a nongiant cell variant of giant cell aortitis and nonspecific aortitis. Takayasu's aortitis is unlikely in a person of this age. A luetic aneurysm is unlikely due to the negative Warthin–Starry stain, with appropriate serologic testing being negative.
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Fig. 1. (A) Ascending aortic aneurysm with adventitial thickening and periaortic adhesions. (B) Thickened and calcified aneurysmal wall.
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Fig. 2. (A) Thickened aortic wall up to 0.9 cm. The luminal surface had multiple foci of atherosclerotic plaques. No gross dissection was identified. (B) 100x Magnification. A lymphoplasmacytic infiltrate is seen centered on the vasa vasorum with germinal center formation. The inflammation extends into the muscular media (lower left). The intima of the small arterioles is thickened. The adventitia is thickened and fibrotic (upper right).
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The etiology of inflammatory aortic aneurysms is multifactorial. Hereditary, environmental and infectious factors all play an important role. It may be related to a process of chronic periarteritis, idiopathic mediastinal fibrosis or perianeurysmal retroperitoneal fibrosis. The inflammatory response in these disorders could be because of an autoimmune response to a component of the atherosclerotic plaque [4]. Our patient's history of idiopathic retroperitoneal fibrosis supports this theory. The cause of inflammatory disease of the aorta is multifactorial. Hereditary, environmental and infectious factors all play an important role. The role of viral agents in the development of inflammatory aortic aneurysms has also been postulated [10]. The reason why the ascending and descending thoracic aorta is so rarely involved remains unclear.
Although the natural history of inflammatory aneurysms of the ascending aorta is unknown, it is believed that they behave similar to the infrarenal abdominal aneurysms. Like any ascending aortic aneurysm, potential complications include rupture, dissection and superior vena cava syndrome. Intervention criteria and surgical approach should be similar to other ascending aortic aneurysms.
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Acknowledgments
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Special thanks to Drs L. Maximillian Buja and Dollett T. White for their pathological expertise. We would also like to thank Kirk Soodhalter for his editorial assistance and Chris Akers for his photographic preparation.
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References
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- 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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Circulation,
October 30, 2007;
116(18):
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Abstract
Introduction
