Ann Thorac Surg 2006;81:178-182
© 2006 The Society of Thoracic Surgeons
Original article: Cardiovascular
Surgical Outcome of Aortic Arch Repair for Patients With Takayasu Arteritis
Kaoru Matsuura, MD,
Hitoshi Ogino, MD
*
,
Hitoshi Matsuda, MD,
Kenji Minatoya, MD,
Hiroaki Sasaki, MD,
Toshikatsu Yagihara, MD,
Soichiro Kitamura, MD
National Cardiovascular Center, Suita City, Osaka, Japan
Accepted for publication June 13, 2005.
* Address correspondence to Dr Ogino, Department of Cardiovascular Surgery, National Cardiovascular Center, 5-7-1 Fujishirodai, Suita, Osaka, Japan 565-8565 (Email: hogino{at}hsp.ncvc.go.jp).
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Abstract
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BACKGROUND: Takayasu arteritis can cause segmental dilatation or stenosis of the aorta and its major branches, and surgical treatment of it is still difficult. Our objective was to review late results of aortic arch repair for patients with Takayasu arteritis.
METHODS: Between 1987 and 2003, 21 patients underwent aortic arch repair under circulatory arrest. Diagnosis was performed by pathologic study of specimens for all patients. Total aortic arch repair was performed in 12 patients with separated branched grafts and in 2 patients with the island technique. Selective cerebral perfusion was used in 12 patients and retrograde cerebral perfusion in 2 patients in this type of surgery. Hemiarch replacement using retrograde cerebral perfusion was performed in 7 patients. Craniocervical vascular stenosis was found in 7 patients and aneurysm in 5 patients. The elephant trunk technique was used in 10 patients. The follow-up period was 6.2 ± 4.2 years.
RESULTS: There was one hospital death due to renal failure, and two late deaths, both of which were sudden. Late in follow-up, a patient who had undergone hemiarch replacement 12 years previously required total aortic arch repair for dilatation of the distal arch. Three patients required thoracoabdominal aortic repair and one patient descending aortic repair for residual aortic dilatation late in follow-up. Postoperative spinal infarction occurred in one patient who underwent hemiarch replacement.
CONCLUSIONS: Surgical and late outcomes of aortic arch repair under circulatory arrest appear favorable, though late dilatation of the residual aorta is a matter of concern.
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Introduction
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Takayasu arteritis is known as "pulseless disease," and its typical ophthalmologic symptoms were first described by Takayasu [1]. Its principal pathologic feature is chronic inflammation leading to stenosis, occlusion, or aneurismal degeneration of the aorta and its major branches [2, 3]. Although these conditions sometimes require surgical intervention, surgery for them is difficult because of the serious complications of vascular reconstruction, the most important of which is detachment of the prosthetic graft [4–8]. However, aortic surgery has developed remarkably, and its outcome has improved dramatically [9–11]. For aortic arch lesions, a safe method of cerebral protection has been established and aggressive surgical treatment is now possible with its use. We review late results for consecutive patients with Takayasu arteritis who underwent aortic arch repair to determine whether it is a safe method of surgery for such patients.
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Patients and Methods
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The study population included 21 consecutive patients with Takayasu arteritis who underwent aortic arch repair under circulatory arrest between 1987 and 2003 in our institution. Patients with Behcet disease were excluded. Total aortic arch replacement (TAR) was performed in 14 patients and hemiarch replacement (HAR) in 7 patients. Preoperative and perioperative variables are shown in Tables 1 and 2.
Diagnosis was confirmed by angiography, pathologic examination, or both, using the criteria identified by The American College of Rheumatology [12]. In this series, pathologic diagnosis of Takayasu arteritis was also performed for all patients by intraoperative examination of specimens of the aortic wall. Associated lesions included aortic regurgitation in 14 patients and renal arterial stenosis in two. Twelve patients had cervical arterial lesions (occlusive disease: 8, aneurismal disease: 5, both: 1). These lesions were located in the brachiocephalic artery in 6 patients, common carotid artery in 9 patients, and left subclavian artery in 4 patients. Coronary artery disease, iritis, and pulmonary arterial dilatation were detected in 1 patient each. Clinically significant inflammation was treated by steroid administration preoperatively, and no such inflammation was present at the time of surgery in any patient. Preoperative steroid therapy was performed in 6 patients. Institutional approval of the study was obtained and each patient within the study gave informed consent for serving as a subject.
Operation
The entire operation was performed by median sternotomy under standard anesthesia. Cardiopulmonary bypass was established with arterial cannulation and bicaval drainage. Myocardial protection was achieved using intermittent antegrade cold-blood cardioplegia. For systemic perfusion, only the femoral artery was cannulated in 17 patients, only the ascending aorta in 2 patients, and both the femoral artery and ascending aorta in 1 patient. Cerebral protection for TAR was performed by antegrade selective cerebral perfusion (SCP) in 12 patients and retrograde cerebral perfusion (RCP) in 2 patients. The HAR was performed using SCP in 1 patient and RCP in 6 patients. For SCP, the axillary artery and left common carotid artery were cannulated in 9 patients, the brachiocephalic artery and left common carotid artery in 3 patients, and the brachiocephalic, left common carotid artery, and left subclavian arteries in 1 patient. The TAR was performed by separated grafting technique (ie, grafting of the cervical vessels individually with the quadrifurcated grafts) in 12 patients, and by the island technique in 2 patients. The elephant trunk technique was employed in 10 patients. Teflon felt reinforcement for the aortic wall was used for all anastomoses but two, for which the inclusion technique was used. The patients who underwent surgery with RCP were cooled to a core body temperature of 15 to 18°C, while those who underwent surgery with SCP were cooled to 21 to 28°C. Concomitantly, composite graft aortic root repair was performed in 6 patients, valve sparing aortic root replacement in 3 (David type operation: 2, Yacoub type operation: 1), aortic valve replacement in 1, extra-anatomical bypass in 1, and coronary artery bypass grafting in 1 patient.
Preoperative and Postoperative Steroid Therapy
Preoperatively, patients positive for C-reactive protein (CRP) (value > 1.0) or whose erythrocyte sedimentation rate (ESR) was above 20 mm/hour were placed on steroid therapy until these values returned to within normal range. For patients diagnosed with active-stage inflammation on intraoperative pathologic specimens, we administered steroid therapy postoperatively until CRP and ESR were within normal limits. The dose and duration of preoperative steroid administration were 13.3 ± 9.7 mg (median 15 mg, 5–30) and 8.1 ± 5.9 years (median 5.9 years, 0.5–16), respectively.
Statistical Analysis
All datasets were reviewed retrospectively. All values are expressed as the mean ± SD. Comparative analysis was performed between the patient groups. Differences were examined for significance by univariate analysis (
2 test, two-tailed t test, Fisher's exact test, or Mann-Whitney U test, where appropriate). Values of p less than 0.05 were considered significant. The Kaplan-Meier technique was used to determine survival curves. All statistical analyses were performed using StatView version 5.0 (SAS Institute, Cary, NC).
Results
There was no operative death, but one hospital death occurred. The patient concerned died 11 months postoperatively due to heart, pulmonary, and renal failure. The remaining 20 patients were followed up from 15 months to 16 years (mean, 6.2 ± 4.2 years). Mean length of intubation was 48.8 hours (median 23.5, 12 to 336 hours), mean length of intensive care unit stay 7.35 days (median 4.5, 1 to 41 days), and mean length of postoperative stay 47.4 days (median 37, 17 to 141 days). There were two late sudden deaths. One of the two patients died of rupture of the descending aorta, but cause of the other is unknown. Postoperative steroid administration was required in 6 patients. Actuarial survival curves are shown in Figure 1. The overall survival rate was 82.7% at 10 years. The survival rate at 10 years did not differ between patients who underwent TAR and those who underwent HAR (p > 0.99).
Of the patients who had cervical arterial lesions, 4 underwent HAR and 8 TAR. Diseased lesions could not be excluded in patients who underwent HAR, and in one patient who underwent TAR without quadrifurcated graft, but they could be completely excluded in 5 patients who underwent TAR with quadrifurcated grafts. In the remaining 2 patients who underwent TAR with quadrifurcated graft, diseased lesions could not be excluded because it was too far from the site of anastomosis.
Detachment of the prosthetic graft occurred in none of the patients. Late cardiovascular events occurred in 7 patients: 5 patients required aortic replacement for late dilatation of the residual aorta, and 2 patients required aortic valve replacement for recurrent aortic regurgitation after valve-sparing aortic root replacement. One patient who underwent HAR in 1987 developed aneurismal change of the residual distal arch, and required TAR in 1999. Digital subtraction angiographic films of this patient are shown in Figure 2. Three other patients required thoracoabdominal aortic repair and one required descending aortic repair for dilatation of the residual aorta. Freedom from aortic events is shown in Figure 3.
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Fig 2. (A) Preoperative DSA before HAR in 1987; (B) postoperative DSA performed after HAR in 1987; (C) preoperative DSA prior to redo operation (TAR) in 1999; (D) postoperative DSA after redo operation (TAR) in 1999. (DSA = digital subtraction angiography; HAR = hemiarch repair; TAR = total aortic arch repair.)
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One patient suffered a neurologic event, paraplegia due to spinal infarction, 17 days postoperatively. Two of three patients who underwent valve-sparing aortic root replacement (David: 1, Yacoub: 1) required aortic valve replacement for recurrent AR at a mean time of 49 months after operation.
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Comment
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Takayasu arteritis is a chronic inflammatory disease that induces stenosis, occlusion, and aneurismal change in large arteries [12–16]. It was first reported in 1908 by Takayasu, a Japanese ophthalmologist, as a peculiar type of arteriovenous fistula of the eye [1]. Although this disease was initially thought to be rare and confined to Asian countries, subsequent clinical reports have shown that it occurs worldwide and is more common than initially believed [14].This rare disease is a chronic arteritis affecting the aorta and its major branches including the coronary, carotid, pulmonary, and renal arteries. Patients affected by it sometimes require surgical intervention for occlusive or aneurismal lesions [17]. Its diagnosis is based on the presence of symptoms and signs of ischemic, inflammatory large-vessel disease as well as supportive radiographic findings. The American College of Rheumatology has identified six major criteria for the diagnosis of this arterial disorder [12].
Pathologic specimens of affected arterial walls of patients with Takayasu arteritis reveal a panarteritis characterized by severe thickening of the adventitia, media, and intima [2, 3]. Nasu [2] presented a detailed pathologic review of this disease, in which the main pathologic finding for this disease was found to be severe destruction of the medial elastic fibers that maintain the strength of the aortic wall. In the early and active inflammatory phases, granulomatous inflammation and various types of cellular infiltration are found in the adventitia and the outer part of the media, with marked inflammation of the vasa vasorum. Subsequently, in the scar stage, severe intimal thickening occurs in the areas overlying granulomatous and fibrotic lesions in the media and adventitia. This severe fibrous thickening makes diseased portions of the aorta stenotic or occluded.
Surgical treatment of this disease is difficult, since it is necessary to manipulate fragile and inflamed tissue, and complications such as hemorrhage, pseudoaneurysm, and detachment of the prosthetic graft are often encountered postoperatively. Miyata and colleagues [4] reported the incidence of postoperative anastomotic aneurysm to be 8.5%. In their study, the occurrence of anastomotic aneurysm was not related to the presence of inflammation, preoperative use of steroid, or pathologic stage. However, we have used aggressive postoperative control of inflammation for patients with clinical inflammation in order to prevent this complication. As previous reports from our institution have noted [17], we believe that preoperative steroid administration to improve inflammation as well as its postoperative use to control inflammation may be important. In the present series, 6 patients who required preoperative steroid administration to control clinically significant inflammation underwent postoperative steroid therapy as well. We administered steroid when CRP was positive or ESR increased above 20 mm/hour and until these values returned to within normal range. Appropriate surgical technique is also important. We have invariably used the Teflon felt reinforcement for all anastomoses to avoid subsequent problems with anastomosis especially in patients with Takayasu disease, though we usually use this technique even in routine aortic surgery. As a consequence, in the present series we encountered no pseudoaneurysm and no detachment of the prosthetic graft. It is also important to exclude cervical vascular lesions.
Recently, the outcome of aortic surgery has improved dramatically due to the development of surgical equipment and techniques. In particular, aortic arch surgery under circulatory arrest has recently exhibited considerable development, including refinement of techniques for cerebral protection. Many previous studies have examined cerebral protection and its neurologic outcomes [9, 18–22]. Ueda and colleagues [18] described RCP in detail as a simple and useful method for aortic arch repair. However, they noted that prolonged RCP is a risk factor for postoperative mortality and morbidity. Several authors have reported the advantages of SCP in detail [9, 19–22]. Okita and colleagues [21] reported a prospective comparative study of brain protection for patients undergoing aortic arch repair performed in our institution. They found that neither mortality nor prevalence of early stroke differed between SCP and RCP, though there was a difference in the incidence of transient postoperative neurologic dysfunction between the two techniques. We have recently preferred to use SCP for brain protection because it has been refined and is sophisticated and reliable. We have frequently used axillary arterial cannulation to establish SCP [23]. Strauch and colleagues [24] also used this method, with a success rate of 95%. In addition, Sabik and colleagues [25] reported that axillary artery cannulation is a safe and effective means of providing antegrade arterial flow during cardiopulmonary bypass in patients with severe atherosclerotic or aneurismal disease. For patients with Takayasu arteritis, axillary arterial cannulation is particularly useful. Since systemic cannulation of the ascending aorta is often difficult due to the severe calcification of the aorta in these patients, femoral arterial cannulation has been employed for systemic perfusion. If systemic perfusion is performed through the femoral artery alone, retrograde thromboembolism from the aortic arch or downstream aorta to the cerebral circulation can occur. However, even if femoral cannulation is performed, antegrade perfusion through the axillary artery can avoid this type of thromboembolism. If axillary arterial cannulation is difficult because this artery is thin, the brachiocephalic artery can be used.
Eusanio and colleagues [26] reviewed the surgical outcome of aortic arch repair using a separated grafting technique with quadrifurcated grafts in 230 patients and the island technique in 122 patients. They reported that separated graft anastomosis had no adverse impact on postoperative mortality and morbidity. Okita and colleagues [27] also reported a higher incidence of pseudoaneurysm or residual aneurismal formation after patch repair for a saccular aneurysm of the aortic arch, compared with the separate grafting technique in our institution. We have strongly preferred the separated grafting method for lesions of the aortic arch, especially in patients with Takayasu arteritis. Use of this technique makes it possible to exclude cervical vascular lesions.
The present study was not designed to be a comparative study of HAR and TAR. The operative method that we chose depended on the range of disease in, and the characteristics of, the affected aorta. Whichever we employed, SCP and axillary cannulation were invaluable adjuncts for avoiding aortic cross-clamping. Furthermore, we believe it is better to perform TAR than HAR for patients with cervical vascular lesions, since it is difficult to exclude such lesions with HAR.
This present study yielded important findings concerning late aortic events. A high incidence of such events was observed. This is why we have employed the elephant trunk technique for the future operation, although few redo operations have been necessary so far. It is very important to follow patients closely postoperatively to prevent life-threatening events associated with the residual aorta. The present study also revealed that neurologic complications can occur after aortic arch repair for Takayasu arteritis. Close postoperative follow-up for such complications is also mandatory.
Limitations of this study are that we examined only a small number of patients, that it was performed retrospectively, and that the operations were performed by many surgeons. In addition, endovascular stent-graft placement, which has only recently been developed, could become an alternative approach, though we have not used it yet. The late outcome of surgical treatment of aortic arch repair under circulatory arrest for patients with Takayasu arteritis was favorable, though late dilatation of the residual aorta is a matter of concern.
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Abstract
Introduction
