arterial complications of thoracic outlet syndrome

5
doi:10.1111/j.1744-1633.2006.00289.x Original article Surgical Practice (2006) 10, 52–56 © 2006 College of Surgeons of Hong Kong Blackwell Publishing AsiaMelbourne, AustraliaASHSurgical Practice1744-16252006 Blackwell Publishing Asia Pty LtdMay 20061025256Original ArticleArterial complications of TOSD Singh et al. *Author to whom all correspondence should be addressed. Email: [email protected] Received 13 August 2005; accepted 17 January 2006. Original article Arterial complications of thoracic outlet syndrome Devender Singh,* Rama Krishna Pinjala, Lankala Rama Chander Reddy and Venkat NagLaxmi Vani Department of Vascular Surgery and Endovascular Surgery, Nizam’s Institute of Medical Sciences, Hyderabad, India. Objective: Arterial vascular complications resulting from thoracic outlet compression, although rare, can be substantial and potentially limb threatening. These complications are due to compressions at the thoracic outlet, the treatment of which continues to be a dilemma. The objective of the present study was to review our experience with this problem with particular reference to its management. Methods: We performed a retrospective study of 12 years and retrieved data from the medical records department of Nizam’s Institute of Medical Sciences, Hyderabad, India. A retrospective review identified 35 patients (age range 15–50 years). In 31 patients, the vasculopathy was caused by a cervical rib, soft tissue anomalies (n = 31), and an elongated transverse process ( n = 4). Evaluation included assessment with colour duplex and arteriography with positional maneuvers. Thirty-two patients presented with a fixed pulse deficit, 22 patients had palpable mass and 15 patients had distal embolization. Results: In 31 patients with cervical rib, the rib was excised via a supraclavicular approach, Scalenectomy was performed and the arterial pathology was repaired on its merit, usually by a vein graft replacement or bypass. The elongated process was excised in the other four patients. Twelve patients required thrombectomy of the distal arteries and a bypass procedure (with a vein/prosthetic graft) was performed in 14 patients. Dorsal sympathectomy, as an adjunct, was carried out in 10 patients. In view of their advanced distal disease, four patients were given prostaglandin therapy. Short-term follow up of 2 years showed good results. Conclusion: Our results show that simple excision of the cervical rib with scalenectomy via supraclavicular approach, together with arterial reconstruction (if required) is adequate for arterial vascular complications resulting from thoracic outlet compression. Patients with severe distal disease may require other adjunct procedures like dorsal sympathectomy or prostaglandin infusions along with proximal reconstruction. Key words: arterial thoracic outlet syndrome, cervical rib, prostaglandin therapy, surgery. Introduction The arterial complications of thoracic outlet syndrome (TOS) are unusual, but have undoubtedly have afflicted humans for millennia. The first reported case of TOS appeared in ‘GENESIS’ chapter 22. 1 Huang Ti Nei Ching Sun Wen (the Yellow Emperor’s Classics of Inter- nal Medicine, in 3000 BC) suggested ‘the inner pulses of the arms denote the state of the adjacent regions; they denote the state of the short ribs’. The symptom- atology was first described by Sir Astley Cooper in 1821. 2 John B Murphy of Chicago in 1905 showed a connection between cervical rib and subclavian aneurysm. 3 Arterial damage, causing ischemia of the limb, occurs in less than 5% of all instances of TOS. Arterial complications are usually associated with cervical ribs, or rudimentary first rib, but in 12% have occurred in patients with no osseous abnormality. 4 The physio- pathological factors begin with compression of the subclavian artery which in most patients produces stenosis, post-stenotic dilatation, formation of aneu- rysms and mural thrombosis. In other patients, aneu- rysm does not form, but the compression still causes stenosis, intimal injury and mural thrombosis. With either scenario, distal embolization can occur and pro- duces signs and symptoms of ischemia that can be limb threatening and if not diagnosed and treated in a timely fashion may lead to limb loss. The diagnosis is based on the characteristic history and a positive stress test. The stress tests commonly performed are hyper abduction, ADSON, costoclavic- ular maneuver or EAST (elevated arm stress test). Duplex scanning and arteriography is necessary for further evaluation and treatment. Unlike neurogenic TOS, in arterial TOS, an early surgical intervention should be considered. Surgical decompression of the

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Page 1: Arterial complications of thoracic outlet syndrome

doi:10.1111/j.1744-1633.2006.00289.x Original article

Surgical Practice (2006) 10, 52–56© 2006 College of Surgeons of Hong Kong

Blackwell Publishing AsiaMelbourne, AustraliaASHSurgical Practice1744-16252006 Blackwell Publishing Asia Pty LtdMay 20061025256Original ArticleArterial complications of TOSD Singh

et al.

*Author to whom all correspondence should be addressed.Email: [email protected] 13 August 2005; accepted 17 January 2006.

Original article

Arterial complications of thoracic outlet syndrome

Devender Singh,* Rama Krishna Pinjala, Lankala Rama Chander Reddy and Venkat NagLaxmi VaniDepartment of Vascular Surgery and Endovascular Surgery, Nizam’s Institute of Medical Sciences, Hyderabad, India.

Objective: Arterial vascular complications resulting from thoracic outlet compression, although rare, can besubstantial and potentially limb threatening. These complications are due to compressions at the thoracic outlet,the treatment of which continues to be a dilemma. The objective of the present study was to review ourexperience with this problem with particular reference to its management.Methods: We performed a retrospective study of 12 years and retrieved data from the medical recordsdepartment of Nizam’s Institute of Medical Sciences, Hyderabad, India. A retrospective review identified 35patients (age range 15–50 years). In 31 patients, the vasculopathy was caused by a cervical rib, soft tissueanomalies (n = 31), and an elongated transverse process (n = 4). Evaluation included assessment with colourduplex and arteriography with positional maneuvers. Thirty-two patients presented with a fixed pulse deficit, 22patients had palpable mass and 15 patients had distal embolization.Results: In 31 patients with cervical rib, the rib was excised via a supraclavicular approach, Scalenectomywas performed and the arterial pathology was repaired on its merit, usually by a vein graft replacement orbypass. The elongated process was excised in the other four patients. Twelve patients required thrombectomyof the distal arteries and a bypass procedure (with a vein/prosthetic graft) was performed in 14 patients. Dorsalsympathectomy, as an adjunct, was carried out in 10 patients. In view of their advanced distal disease, fourpatients were given prostaglandin therapy. Short-term follow up of 2 years showed good results.Conclusion: Our results show that simple excision of the cervical rib with scalenectomy via supraclavicularapproach, together with arterial reconstruction (if required) is adequate for arterial vascular complicationsresulting from thoracic outlet compression. Patients with severe distal disease may require other adjunctprocedures like dorsal sympathectomy or prostaglandin infusions along with proximal reconstruction.

Key words: arterial thoracic outlet syndrome, cervical rib, prostaglandin therapy, surgery.

Introduction

The arterial complications of thoracic outlet syndrome(TOS) are unusual, but have undoubtedly have afflictedhumans for millennia. The first reported case of TOSappeared in ‘GENESIS’ chapter 22.1 Huang Ti NeiChing Sun Wen (the Yellow Emperor’s Classics of Inter-nal Medicine, in 3000 BC) suggested ‘the inner pulsesof the arms denote the state of the adjacent regions;they denote the state of the short ribs’. The symptom-atology was first described by Sir Astley Cooper in1821.2 John B Murphy of Chicago in 1905 showeda connection between cervical rib and subclaviananeurysm.3

Arterial damage, causing ischemia of the limb,occurs in less than 5% of all instances of TOS. Arterial

complications are usually associated with cervical ribs,or rudimentary first rib, but in 12% have occurred inpatients with no osseous abnormality.4 The physio-pathological factors begin with compression of thesubclavian artery which in most patients producesstenosis, post-stenotic dilatation, formation of aneu-rysms and mural thrombosis. In other patients, aneu-rysm does not form, but the compression still causesstenosis, intimal injury and mural thrombosis. Witheither scenario, distal embolization can occur and pro-duces signs and symptoms of ischemia that can belimb threatening and if not diagnosed and treated in atimely fashion may lead to limb loss.

The diagnosis is based on the characteristic historyand a positive stress test. The stress tests commonlyperformed are hyper abduction, ADSON, costoclavic-ular maneuver or EAST (elevated arm stress test).Duplex scanning and arteriography is necessary forfurther evaluation and treatment. Unlike neurogenicTOS, in arterial TOS, an early surgical interventionshould be considered. Surgical decompression of the

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thoracic outlet is achieved by resection or excision ofthe culprit (i.e. cervical ribs, first rib, bands etc.) viavarious approaches. Other ancillary procedures likethrombectomy, bypass, sympathectomy, claviculec-tomy may be required, depending upon the lesions.

Only a few series with large numbers of cases havebeen previously reported, so the surgical strategyremains controversial regarding the optimum mode ofaccess, which bony elements to excise, and the man-agement of the thromboembolic complications. Theobjective of the present study was to review our fairlyextensive experiences with problems with particularreference to their management.

Patients and methods

The records of all patients referred to the vascularservices of the Nizam’s Institute of Medical Sciences,Hyderabad with arterial TOS during the last 12 yearswere called from the medical records department.Thirty-five records were found and analysed. Twenty-two were male and 13 were female (mean age35 years, range from 15 to 50 years). The duration ofsymptoms was from 1 week to 2 years. Most commonsymptomatology was claudication and arm weakness.Absent pulses, pulsatile mass and bruit were also thefeatures of many patients (Table 1). Diagnostic evalu-ation included a thorough history, physical examinationand assessment of occupational and recreationalactivities. In addition to palpation of the pulses andauscultation for bruits at neutral, adduction, abductionexternal rotation and specific provocative positions, allpatients were evaluated with segmental pressure mea-surement of the brachial, radial and ulnar arteries.Arterial photoplethysmographic (PPG) studies of thedigits, duplex ultrasonography scanning of the subcla-vian and axillary arteries, and contrast arteriographywere also performed with the patient in the neutralposition and with positional maneuvers. The artery issaid to be dilated when the diameter is increased bytwice that of the adjacent normal arterial segment,whereas an aneurysm is defined as an increase indiameter greater than twice normal.

Subclavian artery lesions due to compression arefurther divided into three stages as shown below.

Stage 1: Lesions having arterial stenosis and minorpost-stenotic dilatation (n = 3).

Stage 2: Lesions having intrinsic arterial damage usu-ally with subclavian aneurysm formation (n = 10).

Stage 3: Stage 2 lesions with distal thromboemboliccomplications (n = 19).

Chest and cervical spine radiographs were obtainedto define the presence of bony abnormalities such asa cervical rib, anomalous first rib or healed clavicularfracture. Cervical ribs were seen in 31 patients (unilat-eral 22; bilateral nine) and the elongated process infour patients.

Treatment was based on the mechanism of arterialdamage, the degree of ischemia and the nature of anyarterial disease and is described briefly below(Table 2).

Stage 1 lesion: Thoracic outlet decompression withscalenectomy and cervical rib excision. Thoracicoutlet decompression.

Stage 2: Thoracic outlet decompression with aneurys-mectomy and arterial reconstruction.

Stage 3: Stage 2 treatments with additional proceduressuch as thrombectomy and bypasses.

In all the cases our approach was supraclavicular.During the follow up each patient was assessed clin-

ically for symptoms and signs of ischemic disease andthe functional status of the limb was recorded. Anenquiry was also made into the presence or absenceof neurological symptoms. Objective assessment ofthe surgical decompression and arterial repair wascarried out by means of SBP/PVR (segmental bloodpressure/pulse volume recording), photoplethysmogra-phy (PPG) and duplex scanning.

Results

Arterial TOS is very rare and the commonest cause inour setup is presence of a cervical rib. Most of thepatients presented with claudication and weakness ofthe upper limb; more so while doing physical work.Supraclavicular swelling was seen in quite a numberof cases, suggesting subclavian aneurysm/dilatation.Table 1. Clinical presentation of the patients with arterial

complications

Pulse deficit 32Bruit 22Pulsatile mass 22Claudication 32Digital vessel embolization 15Acute upper limb ischemia 4Reynaud’s phenomenon 10

Table 2. Artery lesion at thoracic outlet

Post-stenotic aneurysm 8Dilatation 10Stenosis 4Ulcerated plaque 6Occlusion 12

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After evaualation, all the patients were subjected tosurgical intervention. Our principal surgery was cervi-cal rib excision and scalenectomy via supraclavicularapproach. Four patients had elongated transverse pro-cess, which was also excised. Other ancillary proce-dures were based on the mechanism of the arterialdamage, the degree of ischemia and the nature ofarterial disease (Table 3). For stage 1 lesions (n = 3)only surgical decompression was preferred (C-ribexcision + scalenectomy). For stage 2 lesions (n = 10),surgical decompression with excision of the aneurysmand reconstruction of the subclavian artery was done.In stage 3 lesions (n = 19), along with surgical decom-pression and reconstruction of the subclavian artery,few other procedures were performed for the distaldisease. In 14 patients, end-to-end anastomosis wassufficient, whereas in eight patients vein graft wasrequired. Six patients required polytetrafluoroethylene(PTFE) graft, as the vein was not suitable. Thrombec-tomy of the axillary and brachial artery was done in 12patients. Bypass procedure was done in patients withchronically occluded arterial segment. Subclavian tobrachial (four patients), axillo-distal brachial (twopatients) and brachio-brachial (two patients) bypasseswith a vein graft were done. Claviculectomy was alsodone in eight patients for complete decompression. In10 patients having distal ischemia (with Raynaud’sphenomenon), dorsal sympathectomy was also per-formed along with definitive procedure. Out of 32patients who had pulse deficit on presentation, 28patients had palpable pulse after the surgery. In fourpatients, where distal reconstruction was not possible,and pulse was not palpable after the procedure, wesubjected them to a cycle of prostaglandin E2 infusion,postoperatively (100 µg for 5 days). Two patients weregiven four more cycles (total of five cycles) on amonthly basis.

The most common complication after the surgerywas transient paresethesia, which was seen in 17patients. However, all these patients recovered gradu-ally in due course of time. Two patients developedHorner’s syndrome and took months to recover. Threepatients had subcutaneous collection, which wasdrained. Three patients developed minor infections atthe surgical sites. Four patients continued to have clau-dication and weakness in the postoperative period, dueto non-reconstructable distal disease and were givenprostaglandin infusion cycles. They had relief of symp-toms and were back to work. There was no majormorbidity and no mortality. During a short follow up of2 years, patients with stage 1 and stage 2 lesionsshowed good recovery, whereas in the stage 3 group,two patients with long subclavian-brachial bypassgrafts became occluded and these were later managedmedically.

Discussion

Thoracic outlet syndrome is a family of disorders thataffects the subclavian- axillary neurovascular bundleas it exits the thoracic cage. Many symptoms are dueto compression of the neurogenic and venous struc-tures, and arterial complications at this site are notuncommon. Since Mayo5 and Murphy3 made the firstreports of association between a cervical rib and asubclavian artery aneurysm, the awareness and diag-nosis of arterial injuries of the thoracic outlet at a giventime have paralleled the state of knowledge of thecausative mechanism.6 Subsequently described mech-anisms all result in comparison of the neurovascularbundle by components of the thoracic outlet and shoul-der girdle.7 Anatomical mechanisms of the compres-sion have been found to result from the scalenemuscle, the clavicle and first rib, fibrocartilaginousbands, anomaly of the first rib and clavicle fractures.These diverse components all result in a similar patho-logical condition: compression of the axillo-subclavianneurovascular bundle between the rigid structures.Successful therapy requires precise localization anddefinition of the arterial disease and identification ofthe causative compression elements.

Diagnosis and treatment of this disorder requires ahigh level of suspicion. In patients thought to havethoracic outlet compressions, the use of duplex scanor arteriography, including positional views, helps toestablish the diagnosis. The delay of diagnosis is oftendue to incomplete arteriographic examination of theproximal subclavian artery and its relation to abduc-tion-external rotation or hyper abduction maneuvers.

Arterial injuries of the subclavian artery in the tho-racic outlet are generally produced by osseous

Table 3. Operative management of patients with arterialcomplications

Cervical rib excision + scalenectomy 31Arterial reconstruction

End-to-end anastomosis 15Excision and interposition vein graft 8Excision and interposition PTFE graft 6

Distal bypassesSubclavian-brachial 4Axillo-brachial 2Brachio-brachial 2Thrombectomy 12Dorsal sympathectomy 10Claviculectomy 8Excision of transverse process 4

PTFE, polytetrafluoroethylene.

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abnormalities. In various series, the incidence of cer-vical rib are 70% to 100% in patients presenting withsubclavian artery injury within the thoracic outlet. Inaddition, anomalies of the first rib (as seen in four ofour patients) may produce arterial injuries but areuncommon in most series. With cervical ribs, the sub-clavian artery passes over the bony abnormality, com-presses the inferior aspect of the artery and producesan intimal lesion with or without a post-stenotic dilata-tion. The presence of an anomalous first rib can causesimilar arterial changes but impinges on the superioraspect of the artery. Unfortunately, these patients oftenpresent with distal embolism as the initial symptom.Distal embolism is the most debilitating aspect of thisdisease and is frequently difficult to treat. Uncommonly,a normal anatomical muscular structure may producesignificant arterial injury. Hypertrophy of the anteriorscalene muscle has been associated with arterial com-promise of the subclavian artery and occlusion of supr-aclavicular artery with distal embolization.8–13

The therapeutic approach to the arterial complica-tion of the thoracic outlet syndrome continues to becontroversial. It involves consideration on the one handof the methods for achieving bony decompression andproximal arterial reconstruction (the latter, in turn, gov-erning the method of exposure) and, on the other hand,it involves consideration of the best methods for deal-ing with any additional distal embolic occlusion.14

Resection of the first rib is required only where thereis an anomaly of this rib,15 otherwise most authorsfavour a supraclavicular approach that involves com-plete excision of the cervical rib and additional softtissue compressive elements.16–18

The choice of the arterial reconstruction to be usedis less controversial. In cases of aneurysm, the excesslength of the subclavian artery after removal of thecervical rib is further increased and the resection of theaneurysm and anastomosis can be carried out withoutthe need for graft. If the artery is dilated but not aneu-rysmal, some feel there is no need for arterial interven-tion. However, embolizations from a non-resectedsegment remain a potential risk, so we prefer to openup the dilated portion if an ulcerated plaque with muralthrombus is found, then an arterial resection can becarried out with end-to-end anastomosis. If the intimaappears to be healthy, arteriotomy should be closedwith deep bites. If the artery is stenosed, either resec-tion and end-to-end anastomosis or patch angioplastycan be performed. The method of surgical exposureshould be chosen to facilitate the arterial reconstruc-tion and allow adequate bony decompression.19 Asupraclavicular approach is certainly feasible. For firstrib resection, trans axillary approach is much morecomfortable.

Perhaps the most controversial aspect of manage-ment concerns the thromboembolic complications ofthe thoracic outlet syndrome, which will require eitherthrombectomy or bypass procedures or both.

The role of angioplasty and stenting for short seg-ment occlusions remains limited, as most of thesepatients would require definitive treatment of decom-pression of either cervical rib excision or scalenectomyfor the primary pathology in addition to angioplasty andstenting.

Sympathectomy may be of value in some cases ofdistal embolic occlusion. Patients most likely to benefitare those with digital gangrene or trophic changes.

Thrombolytic therapy is an alternative option incases with acute limb-threatening ischemia with oblit-eration of the brachial and forearm vessels. We haveno experience, as all our patients were not suitablefor it.

Prostaglandin therapy seems to be a promisingtreatment, particularly in cases of digital occlusion orwhen proximal reconstruction has been attempted, butstill with residual distal disease. We have achievedgood results in four of our patients; however, thismodality needs larger trials to consolidate its efficacy.

Conclusion

Our experience confirms the classic indications foroperating when TOS with thromboembolic complica-tions is presented. Furthermore, surgery should bedone as soon as stenosis or a post-stenotic dilatationof the subclavian artery appears, to prevent furtherdegradation of the distal vascular bed. The resectionof the disease lesion followed by re-anastomosis isusually feasible. If not, a vein graft should be inter-posed; the surgical approach is supraclavicular. Embo-lic complications require arterial revascularizationproximal to the elbow. Below the elbow, a non-surgicalapproach is preferable, rather than performing a poten-tially hazardous surgery on a damaged distal vascularbed. The role of prostaglandin therapy is encouraging.Last, in our experience, the optimum approach for sur-gical decompression is cervical rib excision with scale-nectomy via supraclavicular approach, as this providesadequate decompression with good long-term results.

References

1. The Holy Bible. King James version. Grand Rapids, MI:Zondervan Publishing House, 1995. Genesis 22: 1–13.

2. Cooper A. On exostosis. In: Cooper A, Cooper B, Travers B(eds). Surgical Essays, 3rd edn. London, 1821; 128.

3. Murphy JB. A case of cervical rib with symptoms resemblingsubclavian aneurysm. Ann. Surg. 1905; 41: 399–406.

4. Judy KL, Heymann RL. Vascular complications of thoracicoutlet syndrome. Am. J. Surg. 1972; 123: 521.

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5. Mayo H. Exostosis of the first rib with strong pulsations ofthe subclavian artery. Lon. Med. Phy. J. (NS) 1831; 11: 40.

6. Machleder HI. Thoracic outlet syndromes: new conceptsfrom a century of discovery. Cardiovasc. Surg. 1994; 2: 137–45.

7. Rob CG, Standeven A. Arterial occlusion complicating tho-racic outlet compression syndrome. Br. Med. J. 1948; 2:709–12.

8. Scher LA, Veith FJ, Haimovici H et al. Staging of arterialcomplications of cervical rib: guidelines for surgical manage-ment. Surgery 1984; 95: 645–9.

9. Haimovici H. Arterial thromboembolism secondary to outletcompression. In: Haimovici H. (ed.). Vascular Surgery: Prin-ciples and Techniques. New York: Appleton-Century-Crofts,1984; 903–10.

10. Heyden B, Vollmar J. Thoracic outlet syndrome with vascularcomplications. J. Cardiovasc. Surg. 1979; 20: 531–6.

11. Martin J, Gaspard DJ, Johnston PW et al. Vascular manifes-tations of the thoracic outlet syndrome: a surgical urgency.Arch. Surg. 1976; 111: 779–82.

12. Haimovici H. Arterial thromboembolism of the upper extrem-ity associated with thoracic outlet syndrome. J. Cardiovasc.Surg. (Torino) 1982; 23: 214–20.

13. Dorazio RA, Ezzet F. Arterial complications of the thoracicoutlet syndrome. Am. J. Surg. 1979; 138: 246–50.

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15. Roos DB. Transaxillary approach for first rib resection torelieve thoracic outlet syndrome. Ann. Surg. 1966; 163: 354–8.

16. Davies AH, Walton I, Stuart E, Morris PJ. Surgical manage-ment of thoracic outlet compression syndrome. Br. J. Surg.1990; 77: 555–7.

17. Brown SC, Charlesworth D. Results of excision of a cervicalrib in patients with the thoracic outlet syndrome. Br. J. Surg.1988; 75: 431–3.

18. Rai KM, Singh BK, Maudar KK. Supraclavicular first ribresection for treatment of thoracic outlet syndrome. MJAFI1996; 52: 83–6.

19. Hempel GK, Shutze WP, Anderson JF, Bukhari HI. 770consecutive supraclavicular first rib resection for thoracicoutlet syndrome. Ann. Vasc. Surg. 1996; 10: 456–63.