Eur J Cardiothorac Surg 2005;27:520-522
© 2005 Elsevier Science NL
Late thoracic aortic dissecting aneurysm following balloon angioplasty for recoarctation after subclavian flap aortoplasty in childhood—successful surgical repair under circulatory arrest
Madathipat Unnikrishnana,*,
Sanjay Theodorea,
Arun Mathew Petera,
Praveen Kumar Neemab
a Department of Cardio-Vascular and Thoracic Surgery, Sree Chitra Tirunal Institute for Medical Sciences and Technology, Trivandrum, Kerala 695 011, India
b Department of Anesthesiology, Sree Chitra Tirunal Institute for Medical Sciences and Technology, Trivandrum, Kerala 695 011, India
Received 17 September 2004;
received in revised form 3 December 2004;
accepted 17 December 2004.
* Corresponding author. Tel.: +91 47 1252 4463; fax: +91 47 1244 6433. (E-mail: unni{at}sctimst.ac.in).
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Abstract
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Balloon angioplasty is universally accepted presently as the primary therapeutic strategy for recoarctation following surgery during infancy and early childhood. This report concerns a 26-year-old lady with cephalobrachial hypertension on ß-blocker who presented with left sided chest pain since 3 months, having undergone surgery for coarctation in early childhood and balloon angioplasty at 17 years of age. Chest X-ray showed prominent aortic knuckle. CT scan chest showed features of residual coarctation with ‘double-barrelled’ upper thoracic aorta of 5cm diameter. Surgery consisted of interposition graft repair of distal arch and upper thoracic aorta under total circulatory arrest through posterolateral thoracotomy leading to excellent recovery.
Key Words: Aortic recoarctation • Balloon angioplasty • Dissecting thoracic aortic aneurysm • Total circulatory arrest
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1. Introduction
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Although balloon angioplasty comes up as primary option for recoarctation, surgery is currently accepted as the best strategy for native coarctation world over [1]. Concern has been voiced regarding high rate of recoarctation and aneurysm formation after balloon dilation of native aortic coarctation [2]. Aneurysm formation and restenosis rates after BA for recoarctation compare favorably with surgery without the attendant risks of re-do surgery [1].
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2. Case report
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A 26-year-old lady presented with history of left sided chest pain since 3 months and an episode of transient neurological event. She had undergone subclavian flap turn-down angioplasty (SFA) for aortic coarctation at the age of 3 years. Subsequently she was detected to have recoarctation at 17 years of age and underwent BA, which brought down trans-coarctation gradient but resulted in a small intimal tear of no immediate consequence. She was then put on ß-blocker and kept under close medical follow up.
Physical examination showed a young lady in no distress with bounding pulses in right upper limb and neck but feeble left upper and lower limb pulses, with classic brachiofemoral delay. Blood pressure was 160/80mmHg in right arm. Transthoracic echocardiography revealed residual coarctation with gradient of 60mmHg and an intimal flap with large false lumen in the upper descending thoracic aorta (DTA). CT scan confirmed double-barrelled aorta, intimal flap beginning at distal arch and ending at level of left hilum with the aneurysm measuring 5cm in diameter. A 4-vessel digital subtraction angiogram visualized normal cerebral vessels, and aortogram revealed an upper DTA aneurysm with significantly narrowed distal aortic arch. Surgical intervention was decided upon in view of chest pain, significant residual coarctation, dissecting aneurysm 5cm in size, and importantly to avoid vascular complications during pregnancy (Fig. 1).
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Fig. 1. (A) CT chest (axial view) showing upper thoracic aortic dissection, arrow indicates the intimal flap. (B) Digital subtraction angiography showing the distal aortic arch narrowing and the descending thoracic aortic aneurysm.
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She was scheduled for surgery under general anesthesia with double lumen endobronchial intubation. Left posterolateral thoracotomy was performed through the previous scar. Cardiopulmonary bypass (CPB) was initiated using cannulation of left femoral artery and vein. Cannulating main pulmonary artery optimized venous drainage. Core cooling was done to 17°C, total circulatory arrest (TCA) achieved; in Trendelenbergs' position distal arch and upper DTA opened and intimal flap was excised. Hemashield graft of 18mm was sutured just beyond left common carotid artery. A clamp was placed 2cm below the proximal suture line and graft cannulated to start antegrade cardio-cerebral perfusion, after 34min of TCA. Distal anastomosis was next completed and whole body perfusion was restarted in 55min. She was weaned off CPB easily and made smooth postoperative recovery but for left iliofemoral venous thrombosis. She left hospital on 10th post-op day on optimal medications. At 3 months follow-up CT scan showed intact repair (Fig. 2).
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Fig. 2. (A) Post operative CT chest showing patent graft and intact repair. (B) Post operative CT reconstruction, arrows indicate the proximal and distal anastomoses.
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3. Comment
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BA has emerged as the preferred treatment of recoarctation after primary surgery [1]. Many groups have used balloon dilation in native coarctation as well, however, long-term follow up has revealed higher rate of recoarctation and aneurysm formation compared to primary surgical therapy [3]. Lower rates of aneurysm formation have been reported after BA for recoarctation [4]. It is postulated that the absence of abnormal tissue as well as presence of fibrous tissue prevents dissection and aneurysm formation in recoarctation [5].
Mechanism of dilation in BA has been noted to result in intimal and medial tears leading to decrease of transcoarctation gradient [6]. Although most of these small intimal flaps heal on their own, there have been isolated reports of DTA aneurysm formation. Workers have studied the pathology of aortic coarctation and found abnormal tissue with lower elastin content and cystic mucoid lakes in autopsy specimens. This was designated as ‘cystic medial necrosis’ that causes an inherent weakness in the coarctation segment [5]. Higher rates of aneurysm formation can be explained by the ‘cystic medial necrosis’ theory that predisposes this area to dissection and aneurysm formation following BA. In patients who have undergone SFA, residual abnormal coarctation tissue is present, which is inherently weak, and dilation can cause dissection and aneurysm formation [7–9]. Surgical management is a challenge as aneurysms are large, adherent to lung, and the whole hemithorax is extremely vascular due to numerous collaterals. It has also been found that there is reduction in the number and size of collaterals if the coarctation is relieved by BA, which puts the patient at risk of paraplegia if a ‘clamp and sew’ technique is employed. ‘Clamp and sew technique’ was however decided against since proximal anastomosis just beyond left common carotid artery ostium was considered hazardous and technically inaccessible without offloading circulation, especially in redo status. CPB with retrograde femoral perfusion, a period of total circulatory arrest (TCA) and antegrade perfusion through a cannula placed in the graft after completion of the proximal anastomosis provided controlled and safe operating conditions. Vagus and recurrent laryngeal nerves were carefully protected since they lay in close vicinity of proximal suture line. Safety of TCA for 34min is well established and we have utilized this strategy for repair of chronic Type III dissection in our practice regularly [10]. Surgical management included resection of all abnormal coarctation tissue and entailed prosthetic graft replacement of the distal arch and upper thoracic aorta. Technique of TCA for repair of postcoarctation native site aneurysm is reported in 21 patients with median time of 33min (range 22–55min) [10]. Our report is in variance in two ways in that post BA dissection after reparative surgery in childhood was the indication and more importantly judicious use of antegrade cardio-cerebral perfusion through graft after completing proximal anastomosis was employed.
Various authors have recorded post-angioplasty dissection maintained on medical management and follow-up. To the best of our knowledge, this is the first report in English literature of life threatening delayed dissecting aneurysm following BA for aortic recoarctation after subclavian flap aortoplasty managed surgically using graft replacement under TCA with successful outcome.
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Acknowledgement
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Authors thank Prof K. Mohandas, Director of the Institute for his kind permission to publish and Prof Kapilamoorthy of Radiology Department for his generous support for pre and postoperative imaging.
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References
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Abstract
1. Introduction
