Ann Thorac Surg 2007;84:1950-1953. doi:10.1016/j.athoracsur.2007.07.025
© 2007 The Society of Thoracic Surgeons
Original Articles: Cardiovascular
Surgical Management of Aortic Regurgitation Associated With Takayasu Arteritis and Other Forms of Aortitis
Osamu Adachi, MD*,
Yoshikatsu Saiki, MD,
Junetsu Akasaka, MD,
Katsuhiko Oda, MD,
Atsushi Iguchi, MD,
Koichi Tabayashi, MD
Department of Cardiovascular Surgery, Tohoku University Graduate School of Medicine, Sendai, Japan
Accepted for publication July 11, 2007.
* Address correspondence to Dr Adachi, Department of Cardiovascular Surgery, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai, 980-8574, Japan (Email: oadachi{at}mail.tains.tohoku.ac.jp).
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Abstract
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Background: Surgical management of aortic regurgitation associated with aortitis can be complicated with occurrence of prosthetic valve detachment or formation of pseudoaneurysm at the suture line. Postoperative morbidity includes progressive dilatation of the aortic root. We sought to assess our midterm and long-term results of surgical management of aortic regurgitation associated with aortitis.
Methods: Between January 1989 and September 2004, 15 patients (17 cases) with aortic regurgitation caused by aortitis were surgically treated. Of 17 cases, 6 cases had aortic valve replacement and 11 cases had aortic root replacement. All the patients were followed up from 3 months to 15 years with a mean follow-up period of 70.8 ± 54.9 months.
Results: There was no hospital death in all the procedures. During the follow-up period, 1 patient died of prosthetic valve detachment, and 2 patients required aortic root replacement for prosthetic valve detachment or aortic root dilatation after aortic valve replacement, whereas there was neither cardiac death nor reoperation after aortic root replacement.
Conclusions: Low operative mortality and favorable long-term outcome may justify lowering the threshold toward aortic root replacement for aortic regurgitation with aortitis in view of the propensity for development of prosthetic valve detachment.
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Introduction
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Aortic regurgitation, a complication often observed in aortitis, results from inflammation of the aortic root and valve. It is a strong predictive factor for prognosis of aortitis. Aortic regurgitation can be managed surgically with aortic valve replacement with a reasonably favorable outcome. However, detachment of the prosthetic valve and pseudoaneurysmal formation at the suture line can occur as devastating complications. Furthermore, there is a risk of dilatation of aortic root after aortic valve replacement because this syndrome is thought to be progressive disease even with anti-inflammatory therapy. We evaluated our surgical results in patients with aortic regurgitation caused by aortitis.
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Patients and Methods
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Patients
This study was approved by the Institutional Review Board of Tohoku University Hospital, and the requirement for informed consent was waived. Between January 1989 and September 2004, 15 patients (17 cases) with aortic regurgitation caused by aortitis were surgically treated at Tohoku University Hospital. There were 2 males and 13 females. The age ranged from 21 to 66 years, with a mean of 44. The diagnosis of aortitis was based on clinical manifestations and postoperative histology examination of the aortic wall. Of these 15 patients, 11 were diagnosed as having Takayasu arteritis, 3 had Behçet disease, and 1 had giant cell arteritis. All except 1 Behçet patient and 1 Takayasu patient were treated with ant-inflammatory drugs including steroids preoperatively. Besides aortic regurgitation, 3 patients had annuloaortic ectasia, 6 patients had marked dilatation of the ascending aorta, 2 patients had coronary artery obstructive disease, and 1 patient had mitral regurgitation.
Surgical Procedures
Aortic valve replacement (AVR) was performed in 6 patients for isolated aortic regurgitation with standard everted mattress sutures intra-annularly. Subcoronary implantation of stentless bioprosthetic valve was done recently in 1 patient. Two patients underwent coronary artery bypass grafting concomitantly.
Aortic root replacement was performed in 11 patients. Of these, 9 patients had aortic regurgitation with annuloaortic ectasia or aneurysm of ascending aorta or both, and 2 had prosthetic valve detachment after AVR. We used a composite graft with mechanical valve in 7 cases, allograft in 3 cases, and stentless bioprosthetic valve in 1 case for aortic root replacement. Concomitant procedures were aortic arch replacement in 3 patients and mitral valve plasty in 1 patient. For reconstruction of aortic root with composite graft, the prosthetic valve and vascular graft were simultaneously placed with pledgeted interrupted sutures at the aortic annulus. When allograft was chosen for aortic root replacement, we buttressed the proximal suture line with autologous pericardium to prevent late aortic dilatation. We performed the coronary button technique for reconstruction of coronary arteries. The distal anastomosis of aorta was also buttressed with autologous pericardium to prevent late pseudoaneurysm at the suture line. Steroid therapy was initiated immediately after surgery.
Statistical Analysis
Categorical variables are expressed as percentages, and continuous variables are expressed as means ± SD throughout the article. Freedom from cardiovascular death or reoperation was analyzed with the method of Kaplan-Meier. Comparisons between groups were made with the log-rank test.
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Results
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The patient’s profile and surgical results are shown in Tables 1 and 2
and Figure 1. There was no hospital death in all the procedures. Follow-up rate was 100%, and the follow-up periods varied from 3 months to 15 years with a mean of 70.8 ± 54.9 months. In terms of mortality, 1 Behçet patient died of prosthetic valve detachment 5 months after AVR and that was likely to be associated with suboptimal anti-inflammatory drug therapy. Another Behçet patient in the aortic root replacement group died of multiple organ failure owing to exacerbation of the systemic Behçet syndrome 34 months after the aortic root replacement.
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Fig 1. Surgical outcomes for 15 patients with aortic regurgitation (AR) associated with aortitis. (AVR = aortic valve replacement.)
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All the patients but the above 1 Behçet patient who died of multiple organ failure have survived until the time point of observation. Neither prosthesis valve detachment nor pseudoaneurysm was observed after aortic root replacement. As far as morbidity is concerned, 1 female patient with Takayasu arteritis had prosthetic valve detachment 7 years after AVR. She was successfully treated with aortic root replacement using a composite graft. Another Takayasu patient developed aortic root dilatation 12 years after AVR and subsequently underwent aortic root replacement with a composite graft.
On the other hand, not only Takayasu patients but also other aortitis patients who underwent aortic root replacement have never had reoperation. Freedom from cardiovascular death or reoperation after AVR at 5 and 10 years was 89% ± 11% and 74% ± 16%, respectively, whereas those after aortic root replacement were 100% and 100%, respectively (Fig 2). Kaplan-Meier curves for freedom from cardiovascular death or reoperation revealed that the patients after AVR had significantly increased risk compared with the patients after aortic root replacement (p < 0.01).
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Fig 2. Comparison of freedom from cardiovascular death or reoperation between the two groups. The aortic valve replacement (AVR) group (dashed line; n = 6) had significantly higher risk for those events compared with the aortic root replacement group (solid line) by logistic regression analysis (p < 0.01).
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Comment
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Aortitis is literally inflammation of the aorta, and it is representative of a cluster of large-vessel diseases that have various or unknown etiologies. In addition to Takayasu arteritis, several nonbacterial, inflammatory diseases, such as Behçet disease, giant cell or granulomatous aortitis, and rheumatoid aortitis, also invove the aorta, its branches, and aortic valve [1–3]. Aortic regurgitation is seen in 13% to 44% of patients with aortitis [4]. In these patients, the inflammatory process involving the ascending aorta and aortic valve principally causes dilatation of aortic annulus and retraction of aortic cusps with resultant uncoaptation [5–7].
In the surgical management of aortic regurgitation in aortitis, the most serious complications are detachment of the prosthetic valve and pseudoaneurysmal formation at the suture line [3, 8–11]. These complications are thought to be due to fragility of the aortic wall along with aortic annular tissue, and refractory inflammation even with anti-inflammatory therapy. In this retrospective study, prosthetic valve detachment was observed in 33% (2 of 6) of the patients after AVR. One patient (no. 5) who suffered from Behçet disease with active inflammatory findings had valve detachment 5 months after AVR. Control of inflammation is crucial to prevent such complication [12, 13]. However, absence of inflammatory signs does not necessarily preclude the risk of valve detachment. In fact, 1 Takayasu patient (no. 2) who had been well controlled with anti-inflammatory drugs before AVR had valve detachment 7 years after AVR. She was well controlled with anti-inflammatory drugs after the first surgery, and the wall of aortic root obtained during reoperation showed no inflammatory changes in the histologic examination. In such cases, long-term steroid therapy might conversely make the aortic annulus fragile.
In contrast to the AVR group, the patients who underwent aortic root replacement had neither valve detachment nor pseudoaneurysm during the follow-up. Even though the patients who manifested dilatation of aortic root secondary to aortitis were considered to have had severe inflammation at the aortic annulus, there have been no valve detachment in the aortic root replacement cases. Exclusion of the entire diseased wall might be associated with favorable postoperative outcome. With regard to the material for aortic root replacement, we prefer to use cryopreserved allografts, when applicable, in anticipation of exerting less stress on the aortic annulus [14]. Its use is, however, limited by availability of the material. Hence composite graft with mechanical valve has been a standard of choice for this entity. Long-term outcome appears to be satisfactory [15].
Recently, late dilatation of the ascending aorta after AVR was reported in Takayasu patients and giant cell arteritis patients [15, 16]. We experienced 1 Takayasu case (patient no.1) who required aortic root replacement because of aortic root dilatation 12 years after AVR. She had been well controlled with anti-inflammatory therapy and her laboratory check-up had consistently revealed negative after the initial operation. The histologic examination of the aortic wall obtained during the second operation revealed no evidence of active inflammation. Instead, the rupture of elastic fibers in the media and thick fibrosis of the adventitia were observed. These findings are supposed to be the sequel stage of aortitis after regression of inflammation. Careful monitoring of the aortic root is mandatory for possible dilatation in spite of regression of inflammation with anti-inflammatory therapy even after AVR.
Although it may be cautious to lump Takayasu arteritis, Behçet arteritis, and giant cell arteritis together, these are systemic nonbacterial inflammatory disorders and are sometimes refractory to anti-inflammatory therapy. Therefore, it could be reasonable to exclude the aortic root prophylactically in that the residual aortic wall could be an origin of aneurysm. In this retrospective study, we were unable to determine the threshold diameter at which root replacement is considered for the patients with aortitis. Low operative mortality and favorable long-term outcome may justify lowering the threshold toward aortic root replacement for aortic regurgitation with aortitis.
In conclusion, we have reported our experience of 17 cases with aortitis. Aortic root replacement was safely performed and showed a higher event-free ratio than did AVR for aortic regurgitation with aortitis.
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References
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Ann. Thorac. Surg. 2007 84: 1953-1954.
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Abstract
Introduction
