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Department of Cardiovascular Surgery, National Cardiovascular Center, Osaka, Japan
Accepted for publication July 19, 2007.
* Address correspondence to Dr Ogino, Department of Cardiovascular Surgery, National Cardiovascular Center, 5-7-1, Fujishirodai, Suita, Osaka, 565-8565, Japan (Email: hogino{at}hsp.ncvc.go.jp).
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Abstract |
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Introduction |
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A 50-year-old man was referred to the National Cardiovascular Center in Osaka, Japan, with multiple and repetitive anastomotic pseudoaneurysms. He had undergone aortic valve replacement for aortic regurgitation at another hospital 2 years earlier. One year later, two pseudoaneurysms had developed, one on the previous aortotomy suture line and another in the left ventricle (LV) in the interleaflet triangle between the noncoronary and left coronary cusps.
He underwent a redo procedure in which the latter was closed using a Dacron patch (DuPont, Wilmington, DE) from the outside, and the aortic root was replaced using the Cabrol technique. Two months later, a recurrence of the LV pseudoaneurysm was detected by ultrasound imaging and computed tomography (CT), and the patient was referred to our hospital.
The patient had a history of classic polyarteritis nodosa (PAN), which had been diagnosed 16 years earlier by physical examination and biopsy. He had received corticosteroids; however, a relapse of PAN had been suspected 6 months before the previous redo operation when a low-grade fever developed. It had been controlled by an increase in the dose of corticosteroids.
Laboratory findings on admission to our hospital showed an elevated erythrocyte sedimentation rate (ESR) of 66 mL/h, a C-reactive protein (CRP) level of 6.19 mg/L, and leukocytosis (11,800/µL). Further examinations ruled out an infectious etiology; therefore, we suspected a relapse of PAN and increased the dose of corticosteroids.
Two months later, the ESR and CRP levels decreased to 20 mm/h and 2.32 mg/L, respectively. However, follow-up CT scans revealed a newly developed 74-mm-diameter pseudoaneurysm around the anastomotic site of the ascending aorta as well as two other coronary anastomotic pseudoaneurysms (Fig 1) of the ascending aorta that were attached to the sternum. The patient was scheduled for surgical repair.
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In the Trendelenburg position, the sternum was opened with systemic retrograde venous perfusion through the femoral vein cannula to prevent cerebral embolism. The anastomotic pseudoaneurysm of the ascending aorta attached to the sternum was simultaneously incised. An interesting finding was that its orifice was situated 1 cm distal to the previous suture line. The arch vessels were clamped, and antegrade selective cerebral perfusion was established with perfusion through the right axillary artery and other arch vessels.
The ascending aorta and the heart were roughly dissected. Venous drainage was then shifted through the superior and inferior vena cavae. The LV venting tube was inserted through the right upper pulmonary vein. Cardiac arrest was induced with retrograde cardioplegia. We made the distal aortic stump proximal to the innominate artery transecting the pseudoaneurysm orifice. A woven Dacron graft was anastomosed to this stump by an open aortic technique using selective cerebral perfusion. We exposed the aortic root while incising the previous graft. The implanted mechanical valve, which was partly detached around the commissure between the noncoronary and left coronary cusps, was removed. The anastomotic pseudoaneurysm of the LV was communicating with that of the left coronary artery. The right coronary artery was also detached.
A composite valved-graft, comprising a 28-mm woven Dacron graft (DuPont) and a 25-mm mechanical valve, was sutured to the annular remnant with the reinforcement using outside and inside Teflon felts (DuPont). The coronary arteries were reimplanted using 8-mm tube grafts.
The durations of selective cerebral perfusion, cardiac arrest, and the surgical procedure were 72, 372, and 592 minutes, respectively.
The patient recovered uneventfully, and the postoperative ultrasound imaging and CT scans revealed no residual pseudoaneurysms (Fig 2). Bacterial culture of the specimen was negative. The pathologic examination of anastomotic pseudoaneurysms showed inflammatory cells, including foam cells and eosinophils, but no findings consistent with PAN were observed.
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We examined a few specimens of the pseudoaneurysms, which had been pathologically influenced by the surgical procedure, and observed no findings consistent with PAN. However, the multiplicity and repetition of anastomotic pseudoaneurysms indicate specific causes. An infectious cause was ruled out by the results of culture study. A congenital cause, such as Marfan syndrome or Ehlers-Danlos syndrome, was ruled out by the physical examination. Nonbacterial inflammatory diseases, such as Takayasu arteritis, Behçet disease, giant cell arteritis, rheumatoid arthritis, spondyloarthropathies, and sarcoidosis, were ruled out by the physical examination and imaging studies.
The two possible causes for these multiple and repetitive anastomotic pseudoaneurysms are aortitis due to PAN or idiopathic aortitis. Idiopathic aortitis is a syndrome that requires ruling out all causes of aortitis. According to Rojo-Leyva and colleagues [2], the frequency of idiopathic aortitis in a large surgical cohort was 4.3%, and in the absence of systemic illness, thoracic aorta aneurysm formation was the most common manifestation. Thirty-one percent of their patients had a history of giant cell arteritis, systemic lupus, Wegener granulomatosis, or other disorders, and new aneurysms developed in 17% of their patients during a mean follow-up period of 41.2 months. Corticosteroids might have been effective in their experience because no new aneurysms developed in patients who received this therapy.
The laboratory findings for our patient on admission showed some nonspecific inflammatory changes; however, there were no other clinical manifestations. A relapse of PAN was thus ruled out and idiopathic aortitis with a history of PAN was considered plausible.
In summary, we describe a rare case of multiple and repetitive anastomotic pseudoaneurysms of the ascending aorta, the LV, and the coronary arteries that was associated with PAN.
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This article has been cited by other articles:
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  H. L. Gornik and M. A. Creager Aortitis Circulation, June 10, 2008; 117(23): 3039 - 3051. [Full Text] [PDF]
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