Original Article ■ SUBCLAVIAN ARTERY ANEURYSMS ■

Subclavian Artery Aneurysms

Lazar B. Davidovi´c, Dragan M. Markovi´c, Sinisˇa D. Pejki´c, Natasˇa S. Kovacˇevi´c, Momcˇilo M. Cˇoli´c and Predrag M. –Dori´c, Institute for Cardiovascular Diseases, Clinical Center of Serbia, Belgrade, Yugoslavia.

We report the management of 14 subclavian artery aneurysms (13 true, one false) occurring in seven male and seven female patients (average age, 48 years). The aetiology of the aneurysms included in eight, atherosclerosis in five and infection in one patient. Twelve aneurysms were of extrathoracic location, while two aneurysms were intrathoracic. Symptoms related to subclavian artery aneurysms were present in 11 patients (compression in four, haemorrhage in one, and ischaemia in six patients), whereas three aneurysms were asymptomatic. All aneurysms were treated surgically. The supraclavicular approach was used in 11 cases, and the combined transsternal and supraclavicular approach was used in two cases. After aneurysm resection, the reconstruction was performed with end-to-end anastomosis in five cases and with saphenous or synthetic grafts in eight cases. One infected subclavian artery aneurysm was treated with carotid to axillary saphenous vein bypass after exclusion of the aneurysm. Five associated brachial embolectomies and one bypass from the axillary to the distal brachial artery were performed. In all thoracic outlet syndrome cases, decompression at the thoracic outlet was also performed. There was no operative mortality, and the early patency rate was 100%. The follow-up period was from 6 months to 10 years (mean, 3.92 years). During this period, one patient died of malignancy and one patient required reoperation due to aneurysmal degeneration of the saphenous vein graft. Surgical treatment is recommended for all patients with subclavian artery aneurysms to prevent potential complications. (Asian J Surg 2003;26(1):7–11)

Introduction Patients and methods

Subclavian artery aneurysms (SAAns) are infrequently seen There were seven men and seven women, with a mean age of peripheral aneurysms.1–5 There are cases of intrathoracic 48 years (range, 23–65 yr) who were treated for SAAns at our and extrathoracic subclavian artery involvement.3,6,7 In 1818, Institute. Patient demographic and clinical data are presented Mott reported the first unsuccessful treatment of an SAAn by in the Table. ligation of the innominate artery.6 Halsted was the first to The aetiologies of SAAn included thoracic outlet syndrome successfully combine ligation with resection of an SAAn in (TOS) in eight, atherosclerosis in five and infection following 1892,8 while Muller described seven SAAns treated with an intra-arterial drug injection in one case (Figure 1). Two of endoaneurysmorraphy.9 In 1953, Bahnson reported the first our patients had intrathoracic aneurysms (Figure 2), while 12 successful resection and reconstruction of a SAAn.10 Three patients had extrathoracic aneurysms (Figure 3). Three patients years later, Schein performed the same procedure using had partially thrombosed giant aneurysms, with a mean an arterial homograft for reconstruction.11 We report our diameter of 6.1 cm, presenting as asymptomatic pulsatile experience with the management of SAAns treated at the masses. Of these, two were extrathoracic, and one with a 10-cm Institute for Cardiovascular Diseases, Serbian Clinical Center, diameter was intrathoracic. Belgrade, Yugoslavia. Symptoms related to SAAns were present in 11 patients.

Address reprint requests to Dr. Lazar B. Davidovic´, Institute for Cardiovascular Diseases, Clinical Center of Serbia, 8 Dr K. Todorovic´a Street, Belgrade 11 000, Yugoslavia. E-mail: [email protected] • Date of acceptance: 6th September, 2002

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Table. Clinical manifestations of patients with subclavian artery aneurysms

N Sex Age Aetiology Loc Symptoms Procedure Follow-up Patency

1M54 Ath E Asymptomatic mass Resection + PTFE graft 10 yr Patent 2F37 TOS E Asymptomatic mass Resection + ASVG 4 yr Patent** 3F63 Ath E Compression Resection + PTFE graft 8 yr Patent 4F60 Ath E Compression Resection + PTFE graft 6 yr Patent 5M65 Infection E Haemorrhage Carotid-axillary artery bypass 2 yr Patent* 6F31 TOS E Ischaemia Resection + TT brachial 5 yr Patent 7F34 TOS E Ischaemia Resection + TT brachial embolectomy 4 yr Patent 8F64 TOS E TIA, ischaemia Resection + TT brachial embolectomy 4 yr Patent 9M38 TOS E TIA, ischaemia Resection + PTFE graft, brachial embolectomy 2 yr Patent 10 M 65 Ath I, E Compression Resection + PTFE graft 6 yr Patent 11 F 43 TOS E Asymptomatic mass Resection + TT 1 yr Patent 12 M 65 Ath I Compression Resection, carotid-subclavian bypass (Dacron) 6 mo Patent 13 M 30 TOS E Ischaemia Resection + ASVG brachial embolectomy 1 yr Patent 14 M 23 TOS E Ischaemia Resection + TT; axillary-brachial bypass 6 mo Patent

Loc = localization; Ath = atherosclerosis; TOS = thoracic outlet syndrome; TT = end-to-end anastomosis; I = intrathoracic; E = extrathoracic; ASVG = autologous saphenous vein graft; TIA = transient ischaemic attack; PTFE = polytetrafluoroethylene. *Two years later, the patient died with patent graft. **Four years postoperatively, a repeat operation was performed due to aneurysmal degeneration of the venous graft.

Four patients had symptoms caused by compression or acute aneurysm expansion. The mean aneurysmic diameter in these four cases was 39mm. One patient had dysphagia from oesophageal compression, one had right-sided chest and back pain, and two patients had sensory and motor signs of brachial plexus compression. The patient with the infected aneurysm presented with supraclavicular skin necrosis and bleeding. The other six patients had upper extremity ischaemic symptoms (one had blue finger syndrome and five had acute hand ischaemia) from brachial artery thromboembolism. In two such cases, transitory ischaemic attacks (TIA) with contralateral hemiparesis were associated with hand ischaemia. The reason Figure 1. Infected pseudoaneurysm of the left subclavian artery after a therapeutic intra-arterial injection of a cytostatic drug. was retrograde embolism of the right carotid artery. The diagnosis was established using selective two paragraphs and the Table. Direct subclavian reconstruction and duplex ultrasonography. In two patients with intra- using a polytetrafluoroethylene (PTFE) graft was performed thoracically located aneurysms, computed tomography of the in five patients, while a saphenous vein graft was used in two thorax was also performed. patients. One bypass from the left common carotid artery to All patients were treated surgically. In 12 cases, a supraclavi- distal subclavian artery using a Dacron graft was performed cular approach to the subclavian artery was used, while in two, after resection of the proximal left SAAn (Figures 4 and 5) in a combined transsternal and supraclavicular approach was one patient. After excision and debridement was performed in used. Procedures for all patients are described in the following the patient with the infected aneurysm, an extra-anatomic

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Figure 2. Angiographic appearance of a giant intrathoracic aneurysm Figure 3. Angiogram of an asymptomatic, extrathoracic left subclavian of the right subclavian artery. artery aneurysm caused by thoracic outlet syndrome. carotid to axillary artery bypass using a saphenous vein graft Discussion was performed. In the remaining five cases, after aneurysm resection, arterial continuity was established with an end-to- Most true SAAns are caused by atherosclerotic disease1–4,6,7,12 end anastomosis, which is described below. (five of our cases) and TOS5,13–17 (eight of our cases). It is well In five of eight patients with TOS, after aneurysm resection, an end-to-end anastomosis was performed. In all patients with TOS, a decompression procedure at the thoracic outlet also was performed before the reconstruction (two cervical rib resections, two resections of both the cervical and the first ribs and four first rib resections, through the supraclavicular approach). Five brachial embolectomies in cases complicated with brachial artery embolism were performed. In one such case where embolectomy was not possible, a bypass from the axillary to distal brachial artery, with a saphenous vein graft was performed.

Results Figure 4. After complete resection of a giant right subclavian artery aneurysm using a combined transsternal and supraclavicular The overall postoperative complication rate was 21% (three approach, replacement was performed using a polytetrafluoroethylene cases). There were two pneumothoraces after first rib resection (PTFE) graft. The arrow shows the right common carotid artery. performed for decompression, in cases caused by TOS, and one transient median nerve paresis. Both pneumothoraces were treated by drainage. The early patency rate was 100%. Patients were monitored postoperatively by physical examination, Doppler and duplex ultrasonography at 1-, 3-, 6- and 12-month intervals, and once yearly thereafter. The mean follow-up period was 3.92 years (range, 6 mo to 10 yr). During this time, there were no late occlusions, yielding a long-term patency rate of 100%. One patient died with a patent graft due to malignancy. One late complication was identified, which was partial thrombosis of the saphenous vein graft true aneurysm. This aneurysm was resected and replaced with a PTFE graft. Postoperative histological examination showed a Figure 5. Follow-up angiogram showing a patent polytetrafluoro- ethylene graft after repairing an intrathoracic aneurysm of the right connective tissue disorder of the vein wall. subclavian artery (presented in Figures 2 and 4).

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recognized that TOS may be the leading cause of SAAn. dependent on the aneurysm location.6,7,28,40,41 For right-sided Our eight such cases are one of the largest series. Fibromuscu- intrathoracic SAAns, a median sternotomy is the preferred lar dysplasia,1,18 cystic idiopathic medionecrosis,1,19,20 approach (one of our cases), whereas intrathoracic aneurysms infection1,21,22 and congenital anomalies,23,24 are very rare of the left subclavian artery are best managed through a left causes of true SAAn. Subclavian artery pseudoaneurysms thoracotomy. Nonetheless, in one such situation, we performed result from penetrating5,25 or blunt trauma,27–29, especially in a median sternotomy combined with a left supraclavicular relation to clavicle or first rib fracture.30,31 We had one infected extension. Extrathoracic SAAns can be approached through subclavian artery pseudoaneurysm after intra-arterial injec- supraclavicular incisions. SAAn resection and direct graft tion in a drug addict. Such aneurysms are also described by replacement is the recommended treatment for all extensive other authors.32,33 Other forms of subclavian artery pseu- lesions.2,3,7,12,13 We performed such procedures in seven of doaneurysms include iatrogenic false aneurysms after various our patients, while in one case, a bypass from the common diagnostic34–36 or reconstructive surgical procedures.37,38 carotid artery to the distal subclavian artery was necessary. Atherosclerotic SAAn of intrathoracic location usually Some SAAns caused by TOS allow arterial reconstruction by occur in patients over 60 years of age of either sex, but appear end-to-end anastomosis after aneurysm resection, as were to be more common in men (both of our cases). Extrathoracic possible in five of our cases.15–17 An infected aneurysm (one of SAAns frequently occur in young(< 45yr) female patients. our cases) can be treated with aneurysm exclusion and extra- The common complications of SAAn include compression, anatomic carotid to axillary artery bypass using a saphenous thrombosis, distal embolization and rupture.6,12,39 Com- vein graft.4,22,30 In all cases of associated distal arterial pression or acute expansion of the SAAn can produce other embolization, embolectomy or some other reconstructive symptoms and signs than chest, or shoulder pain, and procedure is indicated. Various endovascular procedures for thus, confuse the diagnosis. There could be respiratory the treatment of subclavian artery pseudoaneurysms are problems because of tracheal compression; dysphagia from described.26,34,43–48 It would be very interesting to compare oesophageal compression; hoarseness from compression of surgical and endovascular treatments of SAAn, though our the right recurrent laryngeal nerve; Horner’s syndrome due to institution does not have any experience with endovascular compression of the stellate ganglion; as well as sensory and surgery. motor signs due to brachial plexus compression.6,12,25,40,41 Four patients with SAAn in our series of patients presented Conclusions with compression symptoms. SAAn rupture can produce intra- or extrathoracic bleeding, Aneurysms of the subclavian artery are an uncommon form including hemoptysis from rupture and erosion into the apex of peripheral arterial aneurysm. Despite their infrequent of the lungs. Subclavian artery pseudoaneurysms (especially occurrence, the potential for serious complications (rupture, infected ones, like the one we had in our series) are more prone thrombosis and embolization) mandates surgical or to rupture than true aneurysms.5,20,26–28 endovascular treatment of all diagnosed SAANs. Most SAAns manifest with upper extremity acute or chronic ischaemic symptoms caused by distal thrombo- References embolization.6,11,12,15–17,31,40 We had six such cases. Neurolo- gical problems from retrograde thromboembolism of the right 1. Mc Collum CH, De Gama AD, Noon GP, DeBakey ME. Aneurysm of carotid or vertebral circulation, are also described.40,42 We the subclavian artery. J Cardiovasc Surg 1979;20:159–64. recognized these manifestations in two of our patients. 2. Wang Z, Yu J, Wang X. Management of subclavian artery aneurysm. Chung Hua Wai Ko Tsa Chin 1996;34:359–60. Preoperative duplex ultrasonography and angiography are 3. Pagni S, Denatale RW, Ferneini AM. Atherosclerotic aneurysm of always mandatory for planning the surgical treatment of an the proximal subclavian artery: a case report. Am Surg 1998;64: 323– extrathoracic SAAn. Nonetheless, in cases of intrathoracic 7. aneurysms, computed tomography or magnetic resonance 4. Nonami Y, Ogoshi S. Intrathoracic aneurysm of the right subclavian imaging scans is necessary.6,7,25,27,29,31 artery. A case report. J Cardiovasc Surg 1998;39:39–42. 5. Rossbach MM, Baptiste RC, Sykes MT, et al. Dual-inflow great vessel Large aneursyms are more prone to cause compression and aneurysms: delayed presentation after penetrating trauma. Ann to rupture while small aneusysms tend to thrombose and Thorac Surg 1997;63:238–40. produce distal embolism. The surgical approach to SAAns is 6. Salo JA, Ala-Kulju K, Heikkinen L, et al. Diagnosis and treatment of

10 ASIAN JOURNAL OF SURGERY VOL 26 • NO 1 • JANUARY 2003 ■ SUBCLAVIAN ARTERY ANEURYSMS ■

subclavian artery aneurysms. Eur J Vasc Surg 1990;4:271–4. 29. Schwartz E, Polterauer P. Post-traumatic aneurysm spurium of the 7. Porcellini M, Selvetella L, Scalise E, et al. Arteriosclerotic aneurysms subclavian artery. Unfallchirurg 1996;99: 73–5. [In German] of the subclavian artery. Minerva Cardioangiol 1996;44:433–6. 30. Johnson B, Thursby P. Subclavian artery injury caused by a screw in 8. Halsted WS. Ligation of the first portion of the left subclavian artery a clavicular compression plate. Cardiovasc Surg 1996;4:414–5. and excision of a subclavian-axillary aneurysm. Johns Hopkins Hosp 31. Coulier B, Mairy Y, Etienne PY, Joris JP. Late diagnosis of a traumatic Bull 1892;24:93. pseudoaneurysm of the subclavian artery. J Belge Radiol 1996;79: 9. Muller GP. Subclavian aneurysm with report of a case. Ann Surg 26–8. 1935;101:568. 32. Orteg JL, Neira F, Ruiz C, et al. Intraoperative rupture of subclavian 10. Bahnson HT. Definitive treatment of saccular aneurysm associated aneurysm caused by self intra-arterial injections in a drug addict. Rev with idiopathic cystic medial necrosis. Ann Thorac Surg 1978;26:387. Esp Anestesiol Reanim 1995;42:156. [In Spanish] 11. Schein CP. Arterial thrombosis associated with cervical ribs. Surgical 33. Espirity MB, Medina JE. Complications of heroin injections in the considerations. Surgery 1956;40:428. neck. Laryngoskope 1980;90:1111–3. 12. Dougherty MJ, Calligaro KD, Savarese RP, DeLaurentis DA. 34. Baldwin RT, Kieta DR, Gallagher MW. Complicated right subclavian Atherosclerotic aneurysm of the intrathoracic subclavian artery: a artery pseudoaneurysm after central . Ann Thorac Surg case report and review of the literature. J Vasc Surg 1995;21:521–9. 1996;62:581–2. 13. Mayo H. Exostosis of the first rib with strong pulsations of the 35. Pastores SM, Marin ML, Veith FJ, et al. Endovascular stented graft subclavian artery. Lon Med Phys J (NS) 1831;11:40. repair of a pseudoaneurysm of the subclavian artery caused by 14. Halsted WS. An experimental study of circumscribed dilatation of percutaneous internal jugular vein cannulation: case report. Am J an artery immediately distal to a partially occluding band and its Crit Care 1995;4;472–5. bearing on the dilatation of the subclavian artery observed in certain 36. Feohwein S, Ververis JJ, Marchall JJ. Subclavian artery dissection cases of cervical rib. J Exp Med 1916;24:271. during diagnostic cardiac catheterization: the role of conservative 15. Iida H, Mori H, Mochizuki Y, et al. A case report of thoracic outlet management. Cath Cardiovasc Diagn 1995;34:313–7. syndrome with acute arterial obstruction caused by abnormal first 37. Shmitter SP, Marx M, Bernstein R, et al. -induced rib. Nippon Kyobu Geka Gakkai Zasshi 1997;45:2026–9. subclavian artery dissection in a patient with internal mammary 16. Gruss JD, Geissler C. Aneurysms of the subclavian artery in thoracic artery graft: treatment with endovascular and stent graft. AJR outlet syndrome. Zentralbl Chir 1997;122:730–4. Am J Roentgenol 1995;165:449–51. 17. Nehler MR, Taylor LM Jr, Moneta GL, Porter JM. Upper extremity 38. Valliatu J, Jairaj P, Delamie T, et al. False aneurysm following modified ischemia from subclavian artery aneurysms caused by bony Blalock-Taussig shunt. Thorax 1994;49:383–4. abnormalities of the thoracic outlet. Arch Surg 1997;132:527–32. 39. Bower TC. Aneurysms of the great vessels and their branches. Semin 18. Hirooka S, Oshikiri N, Kimura Sugawara M, et al. A left subclavian Vasc Surg 1996;9:134–46. arterial aneurysm caused by fibromuscular dysplasia: a case report. 40. Clagett GP. Upper Extremity Aneurysms. In: Rutherford RB, ed. Kyobu Geka 1995;48:221–3. Vascu-lar Surgery 5th edition. Philadelphia: WB Saunders, 2000:1356– 19. Persaud V. Subclavian artery aneurysm and idiopathic cystic 69. medionecrosis. Br Heart J 1968;30:436. 41. Sato K, Nonami Y, Yamamoto A, et al. A right subclavian artery 20. Fee HJ, Gerwitz H, O’Connell TX, Grolman JH. Bilateral subclavian aneurysm with hoarseness. Nippon Kyobu Geka Gakkai Zasshi 1995;43: artery aneurysm associated with idiopathic cystic medial necrosis. 567–70. Ann Thorac Surg 1978;26:387–90. 42. Symonds CP. Two cases of thrombosis of subclavian artery with 21. Hara M, Bransford RM. Aneurysm of the subclavian artery associated contralateral hemiplegia of sudden onset, probably embolic. Brain with contiguous pulmonary tuberculosis. J Thorac Cardiovasc Surg 1927;50:259. 1963;46:256. 43. May J, White GH, Yu W, et al. Endoluminal repair: a better option for 22. Law NW, Parvin SD, Darke SG. Diagnostic features and management the treatment of complex false aneurysms. Aust NZ J Surg 1998;68; of bacterial arteritis with false aneurysm formation. Eur J Vasc Surg 29–34. 1994;8:199–204. 44. Sullivan TM, Bacharach JM, Perl J, Gray B. Endovascular management 23. Dobbins WO. Bilateral calcified subclavian arterial aneurysms in a of unusual aneurysms of the axillary and subclavian artery. J Endovasc young adult male. N Engl J Med 1961;265:537. Surg 1996;3:389–95. 24. Rossi PI, Scher LA, Friedman SG, et al. Subclavian artery 45. Gordon MK, Lawrence-Brown MM, Hartley D, et al. A self-expanding pseudoaneurysm in type IV Ehlers-Danlos syndrome. J Vasc Surg endoluminal graft for treatment of aneurysm: results through the 1998;27:549–51. development phase. Aust NZ J Surg 1996;66:621–5. 25. Jebara VA, Oussouldjogli E, Rossi I, et al. Aberrant right subclavian 46. Meyer T, Merkel S, Lang W. Combined operative and endovascular artery aneurysms-a surgical review. J Med Liban 1995;43:157–61. treatment of a post-traumatic embolizing aneurysm of the subclavian 26. Patel AV, Marin ML, Veith FJ, et al. Endovascular graft repair of artery. J Endovasc Surg 1998;5:52–5. penetrating subclavian artery injuries. J Endovasc Surg 1996;3:382–8. 47. Ackroyd R, Singh S, Beard JD, Gains PA. Simultaneous brachial 27. Bechini J, Cuadras P, Tenesa M, et al. Chronic traumatic embolectomy and endoluminal stenting of a subclavian artery pseudoaneurysms of right subclavian artery. Eur Radiol 1998;8:63–5. aneurysm. Eur J Vasc Endovasc Surg 1995;10:248–9. 28. Takeshima S, Hatori N, Uryuda Y, et al. A case report of the subclavian 48. Yamaa K, Yamaguchi K, Takeuchi E, et al. Surgery of aneurysm of the pseudoaneurysms due to blunt chest trauma without fracture. left subclavian artery applying imaged thoracoscopy-a case report. Nippon Kyobu Geka Gakkai Zasshi 1997;45:1884–8. Nippon Kyobu Geka Gakkai Zasshi 1995;43:1012–5.

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