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Ann Thorac Surg 2000;69:1576-1578
© 2000 The Society of Thoracic Surgeons

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Case reports

Upper limb ischemia after subclavian flap aortoplasty: unusual long-term complication

^a Department of Cardiovascular Surgery, Hôpital Broussais, Paris, France

Address reprint requests to Dr Fabiani, Département de Chirurgie Cardio-Vasculaire, Hôpital Broussais, 96, rue Didot, 75674 Paris Cedex 14, France

	Abstract

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Repair of isolated coarctation of the aorta by subclavian flap aortoplasty carries the disadvantage of impaired blood supply to the left arm. However, ligation of branches of the subclavian artery can be tolerated without manifest ischemia of the upper extremity. We report the case of a young man who suffered from left upper extremity ischemia 18 years after initial operation. Treatment consisted of carotid-subclavian bypass with good outcome. The surgical approach of coarctation by subclavian aortoplasty should be reserved for specific cases, and if this procedure is performed, ligation of branches of the subclavian artery should be minimized to increase inflow into the left brachial artery.

	Introduction

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Coarctation of the aorta in children is a congenital aortic narrowing of the upper descending thoracic aorta, adjacent to the site of attachment of the ductus ateriosus, which is sufficiently severe that there is a pressure gradient across the area. The aim of treatment is to abolish the pressure gradient, to restore nonturbulent laminar flow, to allow normal child development, and to avoid long-term deterioration. Subclavian flap aortoplasty is a frequently used surgical approach for coarctation repair in neonates. This procedure has the inherent disadvantage of loss of blood supply to the left arm with the rare but potential sequelae that accompany this complication. We report the case of an 18-year-old man who underwent juxtaductal coarctation treatment by subclavian flap aortoplasty at the age of 2 months and who suffers from recent disabling ischemia of the left upper extremity.

The patient, an 18-year-old man, was admitted to the Cardiovascular Surgery Department of the Broussais Hospital suffering from disabling pain in the left upper extremity after even the slightest effort; the symptoms had worsened over several months.

Surgical treatment of coarctation of the aorta was performed when he was 2 months old. The lesion was localized distal to the left subclavian artery in a juxtaductal position and gave rise to severe arterial hypertension and left ventricular myocardial hypertrophy. Coarctectomy was carried out through a left posterolateral approach. Aortoplasty with left subclavian flap was indicated because of the length of the stenosis. There were no postoperative complications, and regular follow-up showed good progress with complete disappearance of the hypertension, recovery of normal myocardial function, and harmonious development through childhood, with the exception of slight hypotrophy of the left upper extremity.

Unfortunately, from December 1997 onward symptoms of dysesthesia appeared, with numbness of the forearm and the left hand after low-intensity efforts such as writing, or exposure to cold. These symptoms were particularly disabling as the patient is left-handed. Clinical examination showed hypotrophy of the left arm compared with the right (acromiodigital length, left brachial, and antebrachial diameters were 2 cm smaller than the right side) and absent pulses at the axillary, brachial, and radial levels. Left arm systolic blood pressure was 40 mm Hg lower than the right side. Symptoms of subclavian steal syndrome were absent.

Doppler ultrasound showed prevertebral and juxtavertebral occlusion of the left subclavian artery. Arteriography of the supraaortic vessels confirmed the subclavian occlusion (Fig 1). Collateral circulation including the intercostal arteries, thyrocervical trunk, and costocervical system bypassed the proximal subclavian artery. The right vertebral artery was therefore highly developed. No residual aortic stenosis was noted. Severity of symptoms and results of the angiogram led us to carry out surgical repair.

View larger version (138K): [in this window] [in a new window]   Fig 1. Preoperative arteriogram of the supraaortic vessels showing a juxtavertebral occlusion of the subclavian artery. (1 = subclavian artery; 2 = vertebral artery; 3 = cervicointercostal trunk.)

The artery appeared undersized due to severe hypoperfusion. After administration of systemic heparin (50 mg/kg) and lateral clamping of the subclavian artery, an end-to-side anastomosis was performed between an expanded polytetrafluoroethylene (PTFE) graft (5 mm) and the subclavian artery. The graft was then tunneled below the anterior scalenus muscle and the internal jugular vein and above the vagus nerve. Then, an end-to-side anastomosis was realized between the prosthesis and the common carotid artery, which had been dissected already. After declamping, satisfactory blood flow was obtained in the distal subclavian artery. As for all carotid procedures in our unit, a continuous electroencephalogram was performed during carotid cross clamping and mean arterial pressure was maintained above 90 mm Hg.

There were no immediate postoperative complications. Clinically, the upper limb extremity returned to normal temperature and a radial pulse was present. At 1-week follow-up, no significant difference between right and left arm blood pressure was noted (110/80 on the left arm, 120/80 on the right arm). Control Doppler ultrasound and arteriography (Fig 2) showed good graft patency.

View larger version (138K): [in this window] [in a new window]   Fig 2. Control arteriogram of the supraaortic vessels showing good patency of the carotid-subclavian bypass.

	Comment

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After sacrifice of the left subclavian artery, the immediate recovery of inflow into the left brachial artery is ensured by the first and second intercostal arteries, the thyrocervical and costocervical trunks, and the vertebral artery if it is preserved. However, some studies have reported, during exercise testing, symptoms of claudication of the upper extremity, which generally does not require surgical correction [3]. Geiss and colleagues [4] reported one case of gangrene of the upper extremity in a series of 35 cures of coarctation by the technique of aortoplasty with subclavian flap, which occurred in a 2-year-old child who also underwent simultaneous aortic valvuloplasty with cardiopulmonary bypass. Mellgren and associates [5] similarly observed two ischemic complications in a series of 34 patients, one requiring amputation of the arm and the other amputation of the first and third fingers. Van Son and coworkers [3] demonstrated that the existence of claudication of the upper extremity was related directly to the number of branches of the subclavian artery ligated during operation. Some authors have suggested modified techniques of aortoplasty to optimize vascularization of the extremity. Thus, De Mendoça and colleagues [6] and Sharma and colleagues [7] have proposed methods of advancement of the origin of the subclavian artery so as to maintain good perfusion of the left upper extremity. Some groups even suggest systematic carotid reimplantation of the subclavian artery. Furthermore, extensive experience with Blalock-Taussig shunts between the subclavian artery and the pulmonary artery shows that reduction of blood flow in the upper extremity rarely leads to disabling ischemic complications [8].

In our patient, follow-up after initial surgical treatment of the coarctation was favorable. At the age of 18 years the patient experienced a relatively sudden onset of symptoms, reflecting a modification of the arm vascularization. We assume that the distal subclavian artery was preferentially perfused by retrograde flow from the left vertebral artery. Juxtavertebral thrombosis of the subclavian artery was probably responsible for the appearance of symptoms. No obvious signs of vertebral steal were noted because of the dominant character of the contralateral vertebral artery. However, vascular surgical repair was indicated because of the severity of the symptoms.

In our view, bypass was the best approach to treat this case. The length of the subclavian occlusion did not allow transposition of the subclavian artery to the common carotid artery or any endovascular procedure. Our team used to perform supraaortic trunk bypasses with PTFE grafts. Some surgeons prefer autogenous veins, which, to be useful, must have a good caliber. Both types of grafts showed excellent long-term patencies [9].

In conclusion, the debate regarding which surgical procedure is best for repair of coarctation is not resolved. The ideal procedure should relieve the coarctation, should be easy to perform with few complications and a low incidence of restenosis, and should cause no vascular impairment in the left arm. Resection and end-to-end anastomosis seems to be the most appropriate method of repair both anatomically and physiologically. If the subclavian flap aortoplasty technique is chosen, the surgeon should endeavor to preserve the collateral circulation as far as possible to ensure normal functioning of the extremity during childhood and into adult age.

	References

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1. Waldhausen J.A., Nahrwold D.L. Repair of the aorta with subclavian flap. J Thorac Cardiovasc Surg 1966;51:532-533.[Medline]
2. Lodge F.A., Lamberti J.J., Goodman A.H., et al. Vascular consequences of subclavian artery transection for the treatment of congenital heart disease. J Thorac Cardiovasc Surg 1983;86:18-23.[Abstract]
3. Van Son J.A.M., Van Asten W.N.J.C., Van Lier H.J.J., et al. Detrimental sequelae on the hemodynamics of the upper limb after subclavian flap angioplasty in infancy. Circulation 1990;81:996-1004.[Abstract/Free Full Text]
4. Geiss D., Williams W.G., Lindsay W.K., Rowe R.D. Upper extremity gangrene. Ann Thorac Surg 1980;30:487-489.[Abstract]
5. Mellgren G., Friberg L.G., Eriksson B.O., Sabel K.G., Mellander M. Neonatal surgery for coarctation of the aorta. Scand J Thorac Cardiovasc Surg 1987;21:193-197.[Medline]
6. De Mendoça J.T., Carvalho M.R., Costa R.K., Filho E.F. Coarctation of the aorta. J Thorac Cardiovasc Surg 1985;90:445-447.[Abstract]
7. Sharma B.K., Calderon M., Ott D.A. Coarctation repair in neonates with subclavian-sparing advancement flap. Ann Thorac Surg 1992;54:137-141.[Abstract]
8. Zahka K.G., Manolio T.A., Rykiel M.J.F., Abel D.L., Neill C.A., Kidd L. Handgrip strength after the Blalock-Taussig shunt. Clin Cardiol 1988;11:627-629.[Medline]
9. Wittwer T., Wahlers T., Dresler C., Haverich A. Carotid-subclavian bypass for subclavian artery revascularization. Angiology 1998;49:279-287.

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