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J Thorac Cardiovasc Surg 2003;125:570-577
© 2003 The American Association for Thoracic Surgery
Surgery for Acquired Cardiovascular Disease |
From the Departments of Cardiovascular Surgery,a Pediatric Cardiology,b Cardiology,c Heart Institute of Japan, Tokyo Women's Medical University, Tokyo, Japan.
Received for publication April 16, 2002. Revisions requested July 10, 2002; revisions received Aug 4, 2002. Accepted for publication Aug 28, 2002. Address for reprints: Masahiro Endo, MD, Department of Cardiovascular Surgery, Heart Institute of Japan, Tokyo Women's Medical University, 8-1, Kawada, Shinjuku, Tokyo 162-8666, Japan (E-mail: ENDO{at}hij.twmu.ac.jp).
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Abstract |
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Introduction |
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1 reported a case of a peculiar wreath-like arteriovenous anastomosis around the papillae of the ocular fundi in a young woman. During the discussion of Dr Takayasu's presentation, Drs Onishi and Kagoshima presented their experience of 2 patients with similar ocular fundus features, which showed the symptom of impalpable radial arteries. Takayasu arteritis has since been found in young women of Asian extraction and is still recognized as a disease with an unknown cause associated with arteritis predominantly at the aortic trunk, its major branches, and the pulmonary arteries. 2
Coronary artery involvement in Takayasu arteritis was first described by Frovig and Loken 3 in 1951, and coronary artery bypass grafting (CABG) was first performed by Young and colleagues 4 in 1973. The incidence of coronary lesions complicating Takayasu arteritis has been reported as 10.5% in autopsy cases 2 and 9% in patients receiving a clinical diagnosis. 5 Despite this incidence, coronary arterial stenosis is not included in Ishikawa's diagnostic criteria. 6 The inflammatory process in the aorta has been implicated as the cause of stenosis in the coronary ostium, with manifestation of angina pectoris as the first symptom. 7-10 We report the abnormal coronary angiographic findings in patients with Takayasu arteritis and the coronary surgical treatments for these patients.
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Methods |
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The subjects were all Japanese and consisted of 4 male and 27 female patients aged 11 to 63 years (mean age, 41.1 ± 13.2 years). Twenty-nine (93.5%) patients had onset of symptoms or clinical findings at 40 years of age or younger, and 2 (6.5%) patients had unstable angina as the first symptom at 40 years of age or older. Twenty (64.5%) patients were diagnosed by using Ishikawa's definitive criteria. The remaining 11 (35.5%) patients showed no "weakness of radial pulse," but 9 met Ishikawa's minor criteria, and 2 did not meet either Ishikawa's major or minor criteria. In the last 2 patients, B52 or DQ-1 haplotypes were identified, and stenosis in the left main coronary artery (LMCA) ostium and abnormal wall thickening in the ascending aorta were observed at coronary revascularization. These 2 patients were given a diagnosis of Takayasu arteritis on the basis of histologic findings of surgical specimens and specific HLA haplotypes. One of these 2 patients (patient 10 in Table 1) showed "weakness of radial pulse" at 19 years postoperatively. Haplotypes of HLA were determined in 8 patients (Table 1).
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Patients suspected of Bechet syndrome or Sjogren disease were excluded in this study. All patients had negative serologic test results for syphilis and rheumatoid factors. All had no history of Kawasaki disease or clinical findings of pseudoxanthoma elasticum.
Diagnostic angiography
Abnormal coronary angiographic findings were defined as stenosis of greater than 75%, coronary aneurysm, ectasia, and coronary steal phenomenon. Abnormal coronary findings in the 31 patients consisted of 24 stenoses of greater than 75%, 3 coronary artery-bronchial artery anastomoses, 3 aneurysmal coronary ectasia, and 1 combined anastomosis and ectasia. The relationships between the types of abnormal coronary angiographic findings and the clinical signs were analyzed.
Preoperative and postoperative steroids
Preoperative steroids (30-40 mg/d prednisone) were used in 5 patients in the active stage of inflammation (defined as body temperature >38°C, C-reactive protein level >5 mg/dL, and erythrocyte sedimentation rate >50 mm/h). In these patients prednisone was tapered to 10 mg/d until the chronic stage of inflammation in 3 patients, and urgent coronary surgery was performed in 2 patients because of unstable angina during the active stage. Postoperative steroids were used in these 5 patients (5 mg/d prednisone) and in another 3 patients during recrudescence of inflammation.
Surgical treatment of coronary artery
Twenty-three of 24 patients (5 male and 19 female patients, age range, 11-63 years; mean age, 41.0 ± 15.1 years) with coronary artery stenosis underwent coronary surgery. Nine had conventional CABG, 10 had CABG directly to the LMCA, 2 had transaortic endarterectomy, 1 had hybrid CABG (off-pump CABG with stent insertion), and 1 had ostial patch angioplasty. Four patients had conventional CABG and concomitant surgeries, including 1 Bentall operation, 1 aortic valve replacement, 1 extra-anatomic aortic bypass, and 1 bypass from the subclavian artery to the common carotid artery. The postoperative follow-up rate was 100%, and the mean follow-up duration was 9.64 ± 6.86 years. The long-term actuarial survival rate was estimated by using the Kaplan-Meier method. Fistulas and aneurysms were not treated surgically because patients with fistulas had no angina, and aneurysms were diffuse.
During the operations, a piece of the ascending aortic wall near the LMCA in 1 patient and a piece of a hyperplastic intima and medium of coronary artery ostium in 2 patients were resected for histology. These specimens demonstrated granulomatous panarteritis with identifiable giant cells and lymphoplasmacytic infiltration. On the other hand, specimens from the best part of the ascending aorta at the proximal anastomotic site for CABG in 5 patients were without specific changes.
Special surgical techniques used
CABG directly to the LMCA was indicated in patients with left main ostial stenosis without calcification and patients in whom an internal thoracic artery (ITA) could not be used. A piece of saphenous vein (SV) was harvested from the thigh. The best part of the SV graft, that with large diameter and without a valve, was used. Bypass was performed from the posterior right side of the aorta to the LMCA through the transverse sinus.
Transaortic coronary ostial endarterectomy was indicated in patients with localized lesions at the coronary ostium and was performed according to our reported method. 11 First, a percutaneous transluminal coronary angioplasty (PTCA) balloon catheter was inserted into a coronary artery with stenosis, and the stenotic portion was dilated and pulled out. Holding the stenotic portion with a piece of thread, the thick intima was resected piecemeal with a beaver knife. Finally, the remaining thick intima was punched out with a 4-mm Aorta-Punch (Scanlan International, St Paul, Minn).
Patch ostial angioplasty was indicated in patients without calcification at the LMCA and was performed by using the proximal portion of a free right ITA.
Hybrid CABG was indicated in patients in whom CABG with an SV graft was difficult because of a calcified porcelain aorta. Hybrid CABG consisted of preoperative stent insertion to the right coronary artery, off-pump CABG to the left anterior descending artery with an ITA, and bypass from the left subclavian artery to the left common carotid artery. Postoperative stenting was performed in the left circumflex artery.
Postoperative catheterization
All patients were routinely scheduled for postoperative catheterization, and 22 patients underwent examination.
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Results |
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Coronary steal phenomenon
Four female patients with a diagnosis of typical Takayasu arteritis had coronary steal syndrome (Table 3). When contrast medium was injected into the coronary artery, the flow was in the direction of the pulmonary bed area. All patients had multiple occlusive pulmonary arteries associated with pulmonary hypertension and a systolic blood pressure of greater than 60 mm Hg.
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Patient 2 (Figure 2) was a 46-year-old woman with fusiform-shaped dilatation of both coronary arteries, with the largest internal diameter being 25 mm. She had mild atypical coarctation but no chest pain. She died of pneumonia at 66 years of age.
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Isolated coronary surgery
Nineteen patients had isolated coronary revascularization, including 6 cases of conventional CABG with 14 distal anastomoses, 10 cases of CABG directly to the LMCA with 3 supplemental conventional distal anastomoses, 2 cases of transaortic endarterectomy, and case of 1 patch ostioplasty. For conventional CABG with SV or ITA grafts, 17 distal anastomoses had graft flows ranging from 20 to 110 mL/min (mean, 80.2 ± 23.4 mL/min). The graft flow of 10 patients receiving CABG directly to the LMCA ranged from 80 to 340 mL/min (mean, 189.7 ± 83.3 mL/min). The graft flow of conventional CABG was significantly lower than that of CABG directly to the LMCA (P = .0014).
The early patency rate was 100% (29/29), including transaortic endarterectomy and patch ostioplasty. One case of anastomotic stenosis required PTCA and was dilated successfully. One patient who had an uneventful perioperative course died of ventricular fibrillation during intensive care unit stay, and the other 18 were uneventful.
Patient 3 (19-year-old woman in 1981) was in Canadian Cardiovascular Society class 4 and had a 99% stenosis at the left main ostium with good collaterals from the right coronary artery (Figure 3, A and B). Transaortic endarterectomy was performed. Postoperative angiography showed a widely patent left main ostium (Figure 3, C and D). She has had no chest pain since the operation.
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The overall early graft patency in the coronary artery position was 85.7% (6/7), with one SV graft occlusion.
Late results
Late results are shown in Figure 4. There were 3 late deaths: 1 sudden death (3.1 years after the operation), 1 cardiac death caused by congestive heart failure (5.5 years after the operation), and 1 noncardiac death caused by ileus (11.4 years after the operation). Re-CABG was performed in 2 patients: 1 patient with conventional CABG after 4 months and 1 with CABG directly to the LMCA after 13.5 years. The actuarial survival rate, including in-hospital deaths, was 86.5% ± 7.3% at 5 years and 81.4% ± 8.4% at 10 years. The cardiac event-free rate (free from cardiac death, re-CABG, PTCA, and new myocardial infarction) was 77.8% ± 8.8% at 5 years and 72.6% ± 9.6% at 10 years. Two patients with transaortic coronary ostial endarterectomy at 20 and 13 years and 1 patient with patch ostial angioplasty at 7 years had uneventful postoperative courses without symptoms.
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Discussion |
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12 Nasu 2 reported the incidence of coronary lesions complicating Takayasu arteritis to be 10.5% (8/76 autopsy cases). On the other hand, Nagata 12 observed cardiac lesions in 91.5%: cardiomegaly in 81.7%, aortic regurgitation in 14.6%, and myocardial infarction in 12.2% of 82 autopsy cases. Ishikawa 6 has established certain criteria for the diagnosis of Takayasu arteritis, but coronary-related lesions are not included. Before 1977, occlusive coronary artery lesions were found occasionally in autopsy cases. 2,5 More recently, complicating coronary lesions were increasingly observed and reported in 45% of 82 autopsy cases of Takayasu arteritis, 12 and 73% of occlusive coronary lesions were localized around the coronary ostium according to the review by Amano and Suzuki. 13 Coronary ostial stenosis occurs as a result of the extension of inflammation-induced intimal proliferation and fibrous contraction from the ascending aorta and the coronary ostia. 12
Patients with left main ostial stenosis complicating Takayasu arteritis require surgical treatment. The 3 surgical options are CABG, surgical angioplasty of the LMCA, and transaortic coronary ostial endarterectomy. The timing of the operation is important, and surgical treatment should be avoided during the active stage of inflammation in Takayasu arteritis. However, when patients have unstable angina, surgical treatment must be performed without delay because one of the major causes of death among patients with Takayasu arteritis is myocardial infarction. 12
Takayasu arteritis is a disease of unknown cause affecting the aorta and its main branches segmentally. 5 The in situ ITA should not be used as a graft for coronary revascularization because Takayasu arteritis is often accompanied by occlusion of subclavian arteries, even though it is well known that the long-term patency of the ITA graft is significantly higher than that of the SV graft in atherosclerotic patients. 14 In patients with Takayasu arteritis, whose occlusive lesion of the subclavian artery is located more distal than the derivation of the ITA, the use of ITA grafts for coronary revascularization might be possible.
In a safety-first policy, conventional CABG is recommended. Sixty-one patients in the review by Amano and Suzuki 13 and 19 patients in the report by Ando and coworkers 15 were treated with myocardial revascularization, and SV grafts were used in 80% of patients in the review by Amano and Suzuki. 13 However, the long-term graft patency with an SV vein graft was limited to 60% at 4 years in the report by Ando and coworkers. 15 In one case we used adjunctive angioplasty rather than complete surgical revascularization, with the aim to improve patency because the diameter of the ITA graft was small and an SV graft is unsatisfactory in long-term patency. Conventional CABG is not the best choice when there is a macroscopic calcification at the ascending aorta.
In conventional CABG for Takayasu arteritis, graft occlusion occurs mainly at the proximal anastomotic site because of intimal thickening of the aorta. 8,15,16 The use of a large-diameter SV graft anastomosed to the LMCA is recommended to obtain a big anastomosis with high flow and to avoid graft occlusion at the proximal anastomosis. However, because of technical difficulties, a few cases of CABG directly to the LMCA have been reported. 13,17
Calcification of the proximal portion of the aorta and involvement of the LMCA might be contraindications for coronary ostioplasty. 18 A piece of autologous pericardium, 19 glutaraldehyde-treated pericardium, 20 and an SV graft 10 have been used as a patch for coronary ostioplasty. We used a piece of the free proximal portion of the ITA as a patch, and there was no intimal hyperplasia. However, the choice of patch material remains unclear because of limited cases.
Transaortic coronary ostial endarterectomy in Takayasu arteritis was first reported by Endo and colleagues 11 in 1982. According to these cases and a review, 21 patients remain free of angina and cardiac events over the long term, and this method can be recommended for young patients. In the report by Ando and coworkers, 15 9 patients who had transaortic endarterectomy were event free up to 4 years. However, perforation, bleeding, or hematoma at the junction of the aorta and the ostium of the coronary artery might occur during the surgical procedures as a result of excessive resection. Long-term follow-up is necessary because most surgical reports only include a few cases, and survival has been prolonged in patients with Takayasu arteritis. 11,15,21
Pulmonary hypertension could influence the morbidity and long-term mortality in Takayasu arteritis, and pulmonary artery involvement has been found in 56% of patients with this disease. 6 Coronary steal phenomenon in Takayasu arteritis is always associated with occlusive pulmonary arteries causing pulmonary hypertension. Surgical indication is suggested when patients have symptoms of angina pectoris caused by coronary steal syndrome. There were 2 surgical reports of patients having angina because of a large connection from the coronary artery to the bronchial artery 22 or to the pulmonary artery. 23 After the connections were ligated or divided surgically, symptoms were relieved, and the electrocardiography findings were normalized. However, the prognosis of patients with coronary steal phenomena is poor because of respiratory distress or right heart failure.
The present cases suggest a relationship between aneurysmal coronary ectasia and severe aortic hypertension of greater than 200 mm Hg, with or without aortic regurgitation, atypical coarctation, and calcified aorta. In a collective review by Matsubara and coworkers, 24 7 autopsy cases of coronary aneurysm complicating Takayasu arteritis were found by 1992, and all were female patients. From our experience, however, ectasia itself is not an indication for surgical intervention.
Takayasu arteritis, with a strong predilection for women and specific geographic areas, has a significant association with HLA specificities. A population study on HLA typing revealed a high frequency of A24, B39, B52, and DQ-1 25,26 in Japanese patients with Takayasu arteritis. These haplotypes of HLA might be helpful in ruling out other types of aortitis.
Lesions associated with Takayasu arteritis are sometimes reversed with steroid therapy. Radial pulse became palpable, and the erythrocyte sedimentation rate and C-reactive protein levels tended to normalize in almost all patients in 2 reported series. 27,28 Iga and associates 29 reported a patient with Takayasu arteritis who had CABG as a result of unstable angina because of narrowing of the left main artery, and regression of ostial stenosis was observed after steroid therapy.
Patients with Takayasu arteritis show 3 types of abnormal coronary findings: coronary aneurysm, coronary steal phenomenon, and coronary ostial stenosis. Surgical treatment is recommended for patients with left coronary ostial stenosis because coronary ischemia can be a major cause of death. CABG directly to the LMCA, transaortic endarterectomy, and patch ostial angioplasty are treatments of choice for ischemia.
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Acknowledgments |
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