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Ann Thorac Surg 2002;73:644-647
© 2002 The Society of Thoracic Surgeons


Case report

Composite graft replacement after aortic valvuloplasty in Takayasu arteritis

Hideyuki Harada, MD*a, Yukiko Honma, MDa, Yoshikazu Hachiro, MDa, Tohru Mawatari, MDa, Tomio Abe, MDb

a Department of Thoracic and Cardiovascular Surgery, Hokkaido Prefectural Kushiro Hospital, Kushiro, Japan
b Department of Thoracic and Cardiovascular Surgery, Sapporo Medical University School of Medicine, Sapporo, Japan

Accepted for publication August 31, 2001.

* Address reprint requests to Dr Harada, Department of Thoracic and Cardiovascular Surgery, Hokkaido Prefectural Kushiro Hospital, 1-4-26 Sakuragaoka, Kushiro-shi, Hokkaido 085-0805, Japan
e-mail: hideyuki.harada{at}pref.hokkaido.jp


    Abstract
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 Abstract
 Introduction
 Comment
 References
 
A 24-year-old woman had undergone valvuloplasty of the aortic valve and external reinforcement of an aneurysm of the ascending aorta during the active phase of Takayasu arteritis 1 year prior to admission to our hospital. On examination, she was diagnosed as having a large false aneurysm of the ascending aorta with annuloaortic ectasia and severe aortic regurgitation, bilateral common carotid artery aneurysms with a left internal carotid artery saccular aneurysm, and bilateral subclavian artery and right vertebral artery obstructions due to Takayasu arteritis. Because of the risk of rupture, surgical intervention was carried out in spite of the fact that aortitis was in the active phase.


    Introduction
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 Abstract
 Introduction
 Comment
 References
 
Takayasu arteritis is an inflammatory vascular disease of unknown etiology that affects the aorta and its main branches, and requires surgical intervention for treatment of occlusive and aneurysmal lesions. There are various problems in the surgical treatment of Takayasu arteritis in the active phase. We report a case in which a repeat of the original surgery was carried out in spite of active-phase aortitis.

A 24-year-old woman who had been diagnosed previously as having Takayasu arteritis based on clinical symptoms and findings such as chest pain and positive C-reactive protein at age 16, was admitted to our hospital in the active inflammatory phase. Steroid therapy was started to control the inflammation. She had undergone valvuloplasty of the aortic valve for grade III aortic regurgitation (AR) and external reinforcement for an ascending aortic aneurysm in the active phase of Takayasu arteritis in another hospital in May 1995, when she had been a student in another prefecture. Although the AR was still grade II after the surgery, she did well and was treated adequately with steroid until 1 year later when she experienced sudden-onset chest pain, shortness of breath on exertion, palpitations, and general malaise, and she presented at our hospital. On admission, her blood pressure was 110/0 mm Hg in the left arm, 120/0 mm Hg in the right arm, and 170/0 mm Hg in both legs. Her pulse was 120/min and regular. She had prominent symmetrical carotid pulsations and bruits, and a grade 3/6 systo-diastolic murmur at the right sternal border. Hemoglobin was 9.7 gm/dL, hematocrit 32.0%, white blood cell count was 8400/mm3, CRP was 4.8 mg/dL, and her 1-hour erythrocyte sedimentation rate was elevated at 66 mm, on steroids. Chest X-ray film showed an enlarged heart (cardiothoracic ratio, 0.70) with signs of congestive heart failure and an extremely large aneurysm of the ascending aorta. An electrocardiogram revealed sinus tachycardia and left ventricular hypertrophy. The echocardiogram also revealed a left ventricular ejection fraction of 0.26 and marked AR. Enhanced computed tomography revealed bilateral common carotid artery aneurysms, a large ascending aortic aneurysm with a transverse diameter of 13 cm, and a descending aortic dilatation. Aortography also revealed a large false aneurysm of the ascending aorta with annuloaortic ectasia and grade IV AR, bilateral common carotid artery aneurysms with a left internal carotid artery saccular aneurysm, and bilateral subclavian artery and right vertebral artery stenoses (Fig 1). Although steroid treatment appeared to be effective in controlling the inflammatory reaction, chest pain continued. Because of the risk of rupture, surgical intervention was carried out in spite of the aortitis being in the active phase.



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Fig 1. Aortogram and drawing showing progressive dilatation of the ascending aorta and arch that led to aneurysmal dilatation and aortic regurgitation.

 
During the surgery, cardiopulmonary bypass was conducted with femoral and right atrial cannulation. The ascending aorta was markedly enlarged (13 cm) and it extended deep into the right thoracic cavity and adhered to the lung. A very thin ejected region of the aneurysmal wall was noted. The ascending aorta was cross-clamped, and antegrade and retrograde cold blood cardioplegia was delivered. The aorta wrapped with old Dacron (C. R. Bard, Haverhill, PA) fabric was opened. A 2-cm defect of the intima, from which the false aneurysm had arisen, was found in the upper part of the previous surgical site. The aortic valve was thin and the aortic valve ring had expanded to 6 cm in diameter. The threads that had been used for the previous valvuloplasty were found in the commissures. The old Dacron fabric, aneurysmal wall, and aortic cusps were extirpated. A 26-mm Gelseal (Sulzer Vascutek Ltd, Renfrewshire, Scotland) Dacron graft, to which a 23-mm Carpentier-Edwards (Baxter, Irvine, CA) pericardial valve had been sewn previously, in anticipation of the possibility of a future pregnancy, was sutured to the aortic annulus. The Carrel patch method was used for reconstruction of the coronary arteries.

When the rectal temperature reached 22°C, hypothermic circulatory arrest was instituted, and the aortic arch aneurysm was opened. The innominate artery, left common carotid artery, and left subclavian artery were cannulated, and selective cerebral perfusion was then instituted. The descending thoracic aorta was completely transected and anastomosed to the 26-mm Gelseal Dacron arch graft with four side limbs in an end-to-end fashion. The graft was clamped, antegrade full perfusion was resumed through the fourth side limb of the arch graft, and proximal graft–graft anastomosis was performed. The ascending aorta was unclamped. The left subclavian artery distal to the stenotic region was reconstructed with the third limb of the arch graft using end-to-end anastomosis. The left internal carotid aneurysm was resected, and anastomosis of the left internal carotid artery to the second limb graft was performed. The left external carotid artery was anastomosed end-to-side to the limb graft. The right subclavian and vertebral arteries were exposed and arteriotomy was performed. The autogenous saphenous vein was connected at the site, and perfused with cold blood. The first limb graft was anastomosed end-to-end to the right common carotid artery just proximal to the bifurcation. Proximal anastomosis between the saphenous vein and the limb graft was created in an end-to-side fashion. Circulation to the head was allowed through the limb grafts. The body temperature was gradually elevated and the heart was defibrillated. The patient was weaned from the bypass without difficulty. The total cardiopulmonary bypass time was 445 minutes, the aortic cross-clamp time was 169 minutes, and the selective cerebral perfusion time was 240 minutes. The postoperative course was uncomplicated. Postoperative aortography revealed smooth blood flow in the ascending aorta and aortic arch (Fig 2). The patient was discharged and has continued to do well on adequate steroid therapy. Pathological examination of the aorta revealed extensive intimal fibrosis and patchy infiltration of inflammatory cells extending from the media to the thickened adventitia, which confirmed the diagnosis of Takayasu arteritis (Fig 3). A segment of the aortic valve was also examined histologically and found to have undergone myxoid degeneration.



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Fig 2. Aortogram and drawing showing the results of the treatment, which consisted of a composite valve graft replacement of the ascending aorta and transverse aortic arch.

 


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Fig 3. Pathological examination of the aorta revealed extensive intimal fibrosis and patchy infiltration of inflammatory cells extending from the media to the thickened adventitia (Hematoxylin and eosin; original magnification, x100).

 

    Comment
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 Abstract
 Introduction
 Comment
 References
 
Although good long-term results for aortic valvuloplasty have been reported [1], we report on repeated surgery after aortic valvuloplasty in a patient with Takayasu arteritis in the active phase. Because, in this case, AR regressed to grade IV in 1 year after the valvuloplasty, and the ascending aorta was markedly enlarged, the patient needed further surgery. At the site of the external reinforcement of the aneurysm, we were unable to prevent expansion of the ascending aorta, and a false aneurysm developed at the interface of the vessel and the Dacron fabric which had been used for wrapping. These complications can prove difficult when external reinforcement [2] and aortic valvuloplasty are undertaken during active inflammatory annuloaortic ectasia.

Because our patient was young and hoped to have a child, we considered performing the aortic valve-sparing operation, which has been reported by David and Feindel [3], and Sarsam and Yacoub [4]. However, a composite graft replacement with a tissue valve was done because the surgery was being repeated due to recurrent aortic regurgitation following valvuloplasty, and there was a possibility of valvular disease such as myxoid degeneration [5]. In fact, myxoid degeneration was confirmed by pathological examination of the removed valve.

An artificial graft with four side limbs was used for reconstruction of the aortic arch, and this approach seems to be effective when there are many anastomotic regions, as in this case. An important surgical consideration in this case was the need for cerebral protection. For this purpose, selective cerebral perfusion with deep hypothermia and three-vessel cannulation and cooling of the surface of the head with ice bags were used. Rewarming was not started until reconstruction of the head vessels had been completed after declamping of the ascending aorta, and cooling of the head was continued. Although an operation should ideally be done after the inflammation has been controlled by steroids [6], we sometimes cannot avoid the need for an operation during steroid treatment in cases where there are findings of aortic regurgitation with cardiac failure and the possibility of aneurysm rupture, as indicated by chest pain, as was seen in this case. This case demonstrates the various vascular lesions of Takayasu arteritis and emphasizes the difficulties of operation during active aortitis. Our patient has done well in the 5 years since the operation was repeated, but we must continue to observe her closely.


    References
 Top
 Abstract
 Introduction
 Comment
 References
 

  1. Cosgrove D.M., Rosenkranz E.R., Hendren W.G., Bartlett J.C., Stewart W.J. Valvuloplasty for aortic insufficiency. J Thorac Cardiovasc Surg 1991;102:571-577.[Abstract]
  2. Dhillon J.S., Randhawa G.K., Straehley C.J., McNamara J.J. Late rupture after Dacron wrapping of aortic aneurysms. Circulation 1986;74(Suppl 1):11.
  3. David T.E., Feindel C.M. An aortic valve-sparing operation for patients with aortic incompetence and aneurysm of the ascending aorta. J Thorac Cardiovasc Surg 1992;103:617-622.[Abstract]
  4. Sarsam M.A.I., Yacoub M. Remodeling of the aortic valve annulus. J Thorac Cardiovasc Surg 1993;105:435-438.[Abstract]
  5. Allen W.M., Matloff J.M., Fishbein M.C. Myxoid degeneration of the aortic valve and isolated severe aortic regurgitation. Am J Cardiol 1985;55:439-444.[Medline]
  6. Isomura T., Hisatomi K., Yanagi I., et al. The surgical treatment of aortic regurgitation secondary to aortitis. Ann Thorac Surg 1988;45:181-185.[Abstract]



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