Ann Thorac Surg 2007;84:1394-1396
© 2007 The Society of Thoracic Surgeons
Case Reports
Rupture of Thoracoabdominal Aortic Aneurysm Associated with Behcet’s Disease
Nobuhiro Umehara, MD,
Satoshi Saito, MD*,
Hikaru Ishii, MD,
Shigeyuki Aomi, MD,
Hiromi Kurosawa, MD
Department of Cardiovascular Surgery, Tokyo Women’s Medical University, Tokyo, Japan
Accepted for publication April 26, 2007.
* Address correspondence to Dr Saito, Department of Cardiovascular Surgery, Tokyo Women’s Medical University, 8-1 kawada-cho, Shinjyuku-ku, Tokyo, 162-8666, Japan (Email: ssaitos{at}hij.twmu.ac.jp).
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Abstract
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Surgical treatment of arterial Behcet’s disease (BD) has a higher incidence of graft-related complications such as anastomotic pseudoaneurysm or graft occlusion. A 64-year-old man presented with a rupture of the thoracoabdominal aortic aneurysms associated with BD. Evaluation shows a large hematoma in the retroperitoneum and multiple aneurysms of the thoracoabdominal aorta. Physical examination and past history fulfills the diagnostic criteria of BD. The abdominal aorta was replaced with an allograft and the major branches were reconstructed with its branches. The postoperative course was uneventful. A 10-month follow-up computed tomographic scan did not show any graft-related complications. This case suggests the usefulness of an allograft for arterial involvement of BD.
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Introduction
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Arterial Behcet’s disease (BD) sometimes induces life-threatening complications such as rupture of the aneurysm and arterial occlusion. Although surgical intervention is frequently required, surgical treatment of arterial BD can be complicated with graft occlusion or anastomotic pseudoaneurysm. We present a case of successfully replaced ruptured thoracoabdominal aortic aneurysm due to Behcet’s disease with cryopreserved abdominal allograft.
A 64-year-old man had suffered from intermittent severe right lower abdominal pain for 1 month. His physical gastrointestinal examination at a nearby hospital did not show any abnormalities. He had an abdominal computed tomographic scan for the persistent dull pain. Dissection of the infrarenal abdominal aorta was found, and he was brought to our institute on the diagnosis of impending rupture of the abdominal aorta. He had history of right uveitis and he went blind at the age of 50 years. He took oral prednisolone (8 mg/day) since then.
On admission, his vital signs were stable. Oral aphthous ulcers and perirectal ulcers were found with careful examination. Moreover a scar of erythema nodosum existed on his left lower limb that was detected. The greatest factor was that he was diagnosed with Behcet’s disease. Laboratory data suggested systemic inflammatory changes (CRP 2.8 mg/dL). HLA type was B52 positive. An abdominal roentgenogram showed intestinal gas and suggested paralytic ileus. A computed tomographic scan showed a large hematoma in the retroperitoneum from the level of the diaphragm to the inferior mesenteric artery and multiple small aneurysms of the thoracoabdominal aorta (Figs 1A,
1B, and 1C). Finally with the diagnosis of a ruptured thoracoabdominal aorta probably due to Behcet‘s disease, an operation was urgently performed.
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Fig 1. (A) Two-dimensional computed tomographic (CT) scan showing the abdominal aorta and large hematoma with positive mantle sign. (B) Two-dimensional CT scan showing the abdominal aorta and hematoma in the retroperitoneum. (C) Three-dimensional CT scan showing multiple small thoracoabdominal aortic aneurysms.
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Under general anesthesia, with the patient in a left lateral position, a spiral skin incision was made. The aneurysm was approached by a left lateral thoracotomy through the eighth intercostal spaces, a diaphragm incision, and through the retroperitoneum. Adhesion around the aorta was severe from the superior mesenteric artery to the bilateral renal artery level. A partial cardiopulmonary bypass was established with femoral artery and vein cannulation. The aorta was cross clamped just above the diaphragm proximally and just above the aortic bifurcation distally. The aneurysm was opened and the aorta ruptured with a 2 cm defect at the level of the renal artery (Fig 2A). The abdominal aorta was replaced with an abdominal allograft provided by Tokyo University Tissue Bank from the level of the tenth thoracic vertebrae to just proximal to the aortic bifurcation. The celiac trunk, superior mesenteric artery, and bilateral renal arteries were reconstructed with the interposition of the small allograft branches (Fig 2B). Weaning from cardiopulmonary bypass was uneventful.
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Fig 2. An arrow shows the defect of the abdominal aorta at the level of (A) the renal artery. (B) An abdominal aorta was replaced with abdominal allograft and major branches of it were reconstructed with the interposition of the allograft branches.
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The postoperative course was uneventful. The patient left the intensive care unit on day 4 after the operation. A follow-up computed tomographic scan on day 16 showed no anastomotic pseudoaneurysm and no graft occlusion. He was discharged from our hospital on postoperative day 21. The use of 8 mg prednisolone was continued.
The recent follow-up computed tomographic scan at 10 months postoperatively has also showed no occlusion or anastomotic pseudoaneurysm.
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Comment
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Bachet’s disease is a multisystem chronic autoimmune inflammatory disease hallmarked by recurrent episodes of oral ulcerations, ocular lesions, ulcerations of genital lesions, and skin lesions [1]. Arterial involvement of BD is rare, but sometimes causes life-threatening complications [1, 2]. Furthermore, when treated surgically, the incidence of graft-related complications such as anastomotic pseudoaneurysm or graft occlusion is high [2]. Previous reports describe that in the case of BD, graft occlusion or anastomotic pseudoaneurysm tend to occur earlier than in other diseases [2, 3]. These make it difficult to select an operative procedure and graft material. Endovascular stent grafting is certainly an attractive option; however, long-term results remain unclear [4].
Histologically BD arteritis is an inflammation of the media and adventitia [5]. The inflammation causes arterial occlusion and aneurysmal formation [3, 6]. Therefore it is possible that this inflammation has some influence on the formation of anastomotic pseudoaneurysm or graft occlusion after surgery. And some reports describe that surgical complication is less frequent under the remission phase of BD than under the active phase [1, 7].
On the other hand, the graft replacement with allograft is an effective surgical option for mycotic aneurysm. In addition, recent reports have also documented the efficacy of the allograft for noninfectious inflammatory cardiovascular disease, including aortic valve regurgitation due to BD [8].
This particular case here fulfills the diagnostic criteria of BD [1], and the aneurysm possesses the characteristics of the presentation of vascular BD. Therefore we selected the graft replacement with cryopreserved allograft for this patient. The computed tomographic scan at the 10-month follow-up did not show any anastomotic pseudoaneurysms, graft occlusions, or recurrence of aneurysm.
In conclusion, graft replacement with cryopreserved allograft for the impending rupture of thoracoabdominal aortic aneurysm due to Behcet’s disease can be an effective surgical option. It is important to continue the remission therapy and carefully follow-up for anastomotic pseudoaneurysm, graft occlusion, and recurrence of aneurysm.
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References
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- Tsuyoshi Sakae, Mitsuhiro Takeno, Noboru Suzuki, Goro Inaba. Behcet’s Disease N Engl J Med 1999;341:1284-1291.[Free Full Text]
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- Hosaka A, Miyata T, Shigematsu H, et al. Long-term outcome after surgical treatment of arterial lesions in Behcet’s disease J Vasc Surg 2005;42:116-121.[Medline]
- Watanabe H, Oda H, Yoshida T, et al. Endovascular stent-grafting for recurrent aneurysm in Behcet’s disease Int Heart J 2005;46:745-749.[Medline]
- Matsumoto T, Uekusa T, Fukuda Y. Vasculo-Behcet’s disease: a pathologic study of eight cases Hum Pathol 1991;22:45-51.[Medline]
- Kobayashi M, Ito M, Nakagawa A, et al. Neutrophil and endothelial cell activation in the vasa vasorum in vasculo-Behcet disease Histopathology 2000;36:362-371.[Medline]
- Kalko Y, Basaran M, Aydin U, et al. The surgical treatment of arterial aneurysms in Behcet’s disease: a report of 16 patients J Vasc Surg 2005;42:673-677.[Medline]
- Sakuma K, Akimoto H, Yokoyama H, et al. Cryopreserved aortic homograft replacement in 3 patients with noninfectious inflammatory vascular disease Jpn J Thorac Cardiovasc Surg 2001;49:652-655.[Medline]
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D. Erer, E. Iriz, V. Y. Imren, and G. L. Oktar
Vascular Surgery in Behcet's Disease
Ann. Thorac. Surg.,
April 1, 2008;
85(4):
1504 - 1505.
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Abstract
Introduction
