Abstract
Background
Current management of axillosubclavian deep venous thrombosis (DVT) often uses thrombolysis for the DVT, prompt first rib removal, and occasional venoplasty or stenting. Our institution has increasingly used anticoagulation alone followed by interval first rib resection. We sought to analyze the effectiveness of this simplified technique.
Methods
Between September 2012 and April 2021, 27 patients were identified within the institution's electronic medical record as having undergone first rib resection for upper extremity DVT. Seven of these patients had undergone preoperative thrombolysis before referral and were excluded. Among the remaining 20 patients, preoperative clinic charts were evaluated for age, venous segment involvement, contralateral limb involvement, presence of documented hypercoagulable state, duration of preoperative and postoperative anticoagulation, and postoperative outcomes.
Results
Of the 20 patients (mean age, 26.2 years; 13 males) presenting with acute axillosubclavian DVT, all patients had right (n = 8) or left (n = 12) arm swelling. Five patients had extremity pain and four had extremity discoloration. Ten had axillosubclavian vein involvement, 9 had subclavian vein involvement, and 1 had axillary vein involvement. Two patients were on oral contraceptives and no patients had any other diagnosed hypercoagulable conditions. The mean duration of preoperative and postoperative anticoagulation was 3.2 ± 2.6 months and 2.1 ± 2.1 months, respectively. Nineteen patients underwent supraclavicular first rib resection and 1 patient underwent transaxillary resection. Twelve patients (60%) demonstrated complete DVT resolution by venous duplex examination during the postoperative period and 8 patients (40%) demonstrated partial recanalization/chronic DVT. Complications included one hemothorax and one thoracic duct injury. All 20 patients remain asymptomatic without arm swelling, with a mean follow-up of 55.1 ± 34.7 months.
Conclusions
Among patients presenting with acute axillosubclavian DVT, anticoagulation alone followed by interval first rib resection proved to be successful in providing symptomatic relief in the short to medium term. By eliminating the need for preoperative thrombolysis and postoperative venograms, this potentially cost-saving algorithm simplifies our management for acute venous thoracic outlet syndrome while maintaining good clinical outcomes. Because this study only analyzed our management algorithm's effectiveness in the short to medium term, the long-term effectiveness of this treatment will need to be demonstrated.
Keywords: Paget-Schroetter syndrome, Axillosubclavian DVT, Anticoagulation, Venous thoracic outlet syndrome, First rib resection
Article Highlights.
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Type of Research: Single-center retrospective observational study
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Key Findings: Treatment for Paget-Schroetter syndrome with preoperative anticoagulation for ≥6 weeks followed by interval first rib resection and postoperative anticoagulation was used to treat 20 patients with venous thoracic outlet syndrome. Post-treatment duplex examination confirmed complete deep venous thrombosis resolution in 60% of patients. All patients remained asymptomatic after 5 years.
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Take Home Message: Substituting anticoagulation alone for thrombolytic therapy before first rib resection for venous thoracic outlet syndrome results in good venous patency and excellent clinical outcomes.
Although most patients with thoracic outlet syndrome (TOS) present with shoulder or arm pain, approximately 5% present with upper extremity deep venous thrombosis (DVT), often termed venous TOS, or effort-related upper extremity DVT. Effort-related axillosubclavian DVT was first described independently by England's Sir Paget (1875) and Germany's Von Schroetter (1884).1,2 Hughes first applied the eponym Paget-Schroetter syndrome (PSS) in 1949, when reviewing 320 patients with effort thrombosis.3 Patients presenting with PSS are often young, physically active, and involved athletically or vocationally in repetitive overhead arm motions.4 Patients with PSS often present with upper extremity swelling, cyanosis, pain, heaviness, and fatigue.5 The diagnosis of PSS is suggested by history and physical examination, and confirmed with noninvasive duplex scanning or either conventional venography, computed tomographic venography, or magnetic resonance imaging.
Treatment for PSS has varied substantially over time. Early treatment included solely arm elevation and anticoagulation.6 By 1985, thrombolysis followed by prompt first rib resection and scalenectomy (FRRS), with subsequent anticoagulation became a common treatment option.6, 7, 8, 9, 10 By 1993, Machleder had convincingly advocated for a treatment algorithm starting with transcatheter thrombolytic therapy, an interval of anticoagulation before first rib resection, and then repeat venography with balloon venoplasty in cases of residual venous stenosis.8,11 This basic algorithm has remained widely used the last several decades.8,9,12
More recently, Guzzo et al13 retrospectively reviewed outcomes after FRRS for patients treated both with and without preoperative thrombolysis. They concluded that preoperative thrombolysis offers no demonstrable benefit over anticoagulation alone in terms of improving venous patency or symptom resolution.13
After the reported analysis by Guzzo et al, our vascular division began to adopt a simplified approach to treating patients presenting acutely with PSS. Those patients presenting at the time of diagnosis were treated for an interval of ≥6 weeks with anticoagulation, followed by FRRS. Postoperatively patients completed their 3- to 6-month course of anticoagulation, and then underwent venous duplex examination. The effectiveness and outcomes of this simplified approach are analyzed here.
Methods
After obtaining approval from our university institutional review board, a retrospective review for all patients with venous TOS from September 2012 to April 2021 was conducted by initially searching our electronic medical record for both Current Procedural Terminology Code for rib resection (21615) and diagnosis code for upper extremity DVT (I82.629). Twenty-seven patients were identified. Seven patients were excluded from the analysis, having been administered preoperative thrombolytics at another hospital before referral.
Among the 20 remaining patients presenting with acute PSS, electronic clinical records were evaluated for patient demographics and comorbidities, symptoms at presentation, venous segment involvement, contralateral limb status, documented hypercoagulable state, anticoagulation duration, surgical approach, and complications. All patients underwent delayed rip resection for documented complete acute axillary and/or subclavian venous occlusion. All patients had presented with arm swelling, five had arm pain, and four had discoloration (Table I).
Table I.
Presenting signs and symptoms among 20 patients with venous thoracic outlet syndrome (TOS)
| Signs and symptoms | No. |
|---|---|
| Pain | 5 |
| Swelling | 20 |
| Discoloration | 4 |
| Bruising | 1 |
Postoperative venous duplex images obtained at the completion of anticoagulation therapy were reviewed and recorded. Short- and medium-term outcomes were tracked including patient symptoms, axillosubclavian vein patency, and venous abnormalities.
Results
Outcomes were reviewed among these 20 patients (13 males, 7 females) presenting to our service with acute effort thrombosis treated without thrombolysis. The cohort averaged 26.2 ± 10.8 years of age (range, 16-53 years) with a mean follow-up of 55.1 ± 34.7 months (range, 8-101 months) (Table II). No patient had a defined hypercoagulable state, although two patients were on previously prescribed oral contraceptives. All patients noted arm swelling at the time of acute presentation, and some had cyanotic discoloration, bruising, or pain (Table I). All 20 patients had complete venous occlusion of an axillosubclavian vein segment. Sonographically, 9 patients had DVTs isolated to the subclavian vein, 1 had DVT isolated to the axillary vein, and 10 had DVTs that extended to include both subclavian and axillary venous segments (Table III). Twelve of the DVTs were in the left arms and none were bilateral. Of the 20 patients studied, 11 had an identifiable effort activity likely related to their DVT development: there were 2 baseball players, 2 dancers, and 1 musician (Table IV).
Table II.
Pre-/post-first rib resection and scalenectomy (FRRS) anticoagulation duration and follow-up
| Characteristics | Months (mean ± SD) |
|---|---|
| Preoperative anticoagulation | 3.2 ± 2.6 |
| Postoperative anticoagulation | 2.1 ± 2.1 |
| Duration of follow-up following FRR | 55.1 ± 34.7 |
Table III.
Individual patient deep venous thrombosis (DVT) treatment and outcomes after first rib resection and scalenectomy (FRRS) among 20 patients listed by age range
| Patient number | Gender | Age range, years | Laterality | Acute DVT venous segment per preoperative imaginga | Anticoagulation duration before FRRS, months | Anticoagulation duration after FRRS, months | Duration of post-FRRS follow-up, months | Final venous duplex: DVT outcome |
|---|---|---|---|---|---|---|---|---|
| 1 | M | 15-20 | Left | Subclavian | 1.5 | 3 | 22 | Complete resolution |
| 2 | M | 15-20 | Right | Subclavian | 1 | 3 | 91 | Chronic with recanalization |
| 3 | M | 15-20 | Right | Subclavian and axillary | 1 | 5 | 97 | Complete resolution |
| 4 | F | 15-20 | Left | Subclavian | 4 | 6 | 95 | Complete resolution |
| 5 | M | 15-20 | Left | Subclavian | 5 | 1 | 88 | Chronic with recanalization |
| 6 | M | 15-20 | Left | Subclavian and axillary | 3 | 0 | 12 | Complete resolution |
| 7 | M | 15-20 | Right | Subclavian, axillary, and basilic | 12 | 0 | 101 | Chronic with recanalization |
| 8 | M | 15-20 | Right | Subclavian and axillary | 1.5 | 3 | 17 | Chronic with recanalization |
| 9 | M | 21-25 | Right | Subclavian | 1.5 | 1.5 | 8 | Chronic with recanalization |
| 10 | F | 21-25 | Right | Subclavian, axillary, and Brachial | 2 | 6 | 85 | Chronic with recanalization |
| 11 | M | 21-25 | Left | Subclavian and axillary | 6 | 0 | 40 | Complete resolution |
| 12 | F | 21-25 | Left | Subclavian, axillary, and cephalic | 1.5 | 1.5 | 24 | Complete resolution |
| 13 | F | 26-30 | Left | Subclavian | 3 | 0 | 37 | Complete resolution |
| 14 | M | 26-30 | Left | Subclavian and axillary | 2 | 4.5 | 90 | Chronic with recanalization |
| 15 | M | 26-30 | Left | Axillary and Brachial | 3 | 0 | 33 | Complete resolution |
| 16 | M | 31-15 | Left | Subclavian | 6 | 3 | 99 | Complete resolution |
| 17 | F | 31-35 | Right | Subclavian | 4 | 0 | 74 | Complete resolution |
| 18 | F | 36-40 | Left | Subclavian and axillary | 1 month | 2 | 21 | Complete resolution |
| 19 | F | 46-50 | Right | Subclavian and axillary | 3 | 0 | 30 | Complete resolution |
| 20 | M | 51-55 | Left | Subclavian | 1 month | 3 | 38 | Complete resolution |
Specific age and effort type not included to preserve patient confidentiality.
Table IV.
Triggering effort activities among 20 patients with venous thoracic outlet syndrome (TOS)
| Activities | No. |
|---|---|
| Sports | 9 |
| Baseball | 2 |
| Basketball | 1 |
| Dance | 2 |
| Soccer | 1 |
| Swimming | 1 |
| Weightlifting | 1 |
| Wrestling | 1 |
| Occupational | 2 |
| Musician | 1 |
| Physical therapist | 1 |
| Unidentified | 9 |
Three patients were anticoagulated with warfarin, nine with rivaroxaban, four with apixaban, and four with enoxaparin. All patients received ≥6 weeks of preoperative anticoagulation. The average duration of preoperative anticoagulation was 3.2 ± 2.6 months and postoperative anticoagulation was 2.1 ± 2.1 months (Table II). All patients had preoperative duplex imaging of their upper extremities to demonstrate the extent of DVT at the time of presentation. All patients had an abnormal preoperative Adson's test on physical examination of the affected extremity.
First rib resection was performed either via the supraclavicular approach (n = 19) or transaxillary approach (n = 1). The surgical approach was based on individual surgeon preference. Division of the rib was performed with a RemB Sagittal oscillating saw (Stryker Instruments, Portage, MI) using a 9-mm blade and foot pedal to maximize costochondral space visualization and anteromedial rib resection. Scalenectomy was partial and limited to that obliged with rib resection. Full decompression of the subclavian vein was confirmed with manual palpation of the final anterior first rib cut edge. This was always confirmed to be well anterior to the palpated and visualized subclavian vein. Operative times varied from 75 to 95 minutes. One patient suffered a hemothorax and one patient suffered a thoracic duct injury.
All 20 patients had resolved their upper extremity swelling by the time of their interval FRRS. Each patient had a normal Adson's test on postoperative physical examination of their affected arm. All arm swelling remained resolved both through the time of anticoagulation completion and the time of data analysis with follow-up of 55.1 ± 34.7 months (range, 8-101 months). All patients have returned to their vocations, avocations, and sports. Twelve patients (60%) demonstrated complete DVT resolution by venous duplex performed at the completion of therapy and 8 patients (40%) demonstrated partial recanalization with either chronic DVT or venous wall thickening.
Discussion
The contemporary treatment of PSS commonly includes (1) anticoagulation to prevent DVT propagation and permit physiologic thrombolysis, (2) catheter-directed thrombolysis or thrombectomy to rapidly decrease thrombus burden and palliate symptoms, and (3) decompression of the thoracic outlet to address root cause. Current treatment algorithms apply 50 years of pharmacologic, endovascular, and surgical advancements. In the historical context detailed elsewhere in this article, the current study sought to analyze PSS patient outcomes using a simplified treatment regimen.
Although upper extremity DVTs were traditionally treated with rest, arm elevation, and anticoagulation, early treatment outcomes cited both high recurrence rates and substantial morbidity.14 In reporting outcomes among 23 conservatively treated patients with PSS in 1965, Adams15 noted a 12% incidence of pulmonary embolism, and a 68% incidence of late residual or recurrent arm pain or swelling. Two years later Coon and Willis16 reported that patients with PSS treated with anticoagulation were less symptomatic than those patients treated with only rest and arm elevation.
In 1971, Adams and DeWeese7 described a cohort of 34 patients with PSS. Although they noted fewer residual or recurrent symptoms among patients treated initially with anticoagulation, disappointingly 70% of their nonoperatively treated patients had residual or recurrent symptoms. They reported results among six patients who underwent surgical thrombectomy for acute localized UE DVT. Five of these six patients underwent concomitant claviculectomy or anterior scalene division. Reporting persistent venous patency among the five patients who underwent simultaneous decompression, they subsequently advocated for “widening of the costoclavicular space” at the time of thrombectomy.
Increasing surgical experience was soon reported with scalenectomy and first rib resection to decompress the costoclavicular space.17,18 By 1985, Taylor et al10 described the successful use of systemic streptokinase followed by elective first rib resection for acute subclavian DVTs. Subsequent authors described catheter directed infusion of streptokinase or urokinase to treat a blended cohort of PSS and secondary upper extremity DVT patients.9
By 1993, Machleder8 reported a series of 50 consecutive patients enrolled in multimodal treatment for PSS, using transcatheter thrombolysis, 3 months of warfarin anticoagulation, transaxillary first rib resection, and postoperative venography with selective angioplasty. Eighty-six percent of the 35 patients who underwent first rib resection remained symptom free through follow-up, and 83% of 27 patients with occluded or recanalized veins after lysis or anticoagulation were symptom free after first rib resection. Notably, more patients were asymptomatic than had normal subclavian venograms, suggesting that good clinical outcomes were often achieved among patients with abnormal final subclavian venograms.
In 2010, Guzzo et al13 reported the results of a retrospective analysis of 103 patients undergoing 110 transaxillary FRRS for PSS. Outcomes among 45 patients undergoing thrombolysis (with or without percutaneous transluminal angioplasty) before FRR were compared with 65 patients simply anticoagulated before FRR. In this latter group of patients presenting to Johns Hopkins, upper extremity DVTs were subacute or chronic at presentation (mean symptom duration, 6.2 months). Interestingly, they reported 91% subclavian venous patency among these patients treated with anticoagulation alone before FRR. Preoperative thrombolysis had little impact on venous patency or symptomatic improvement after FRRS in their large retrospective cohort of patients with PSS.
Given the results reported by Guzzo et al, our surgical division modified its treatment algorithm for patients with PSS encountered at the time of their acute DVT. Patients presenting acutely with PSS were not subjected to thrombolysis, but rather anticoagulated for an interval of ≥6 weeks, after which time they underwent FRRS. After FRRS, they completed 3 to 6 months of anticoagulation (Table II). A minimum interval of 6-weeks of anticoagulation was chosen before FRRS because all patients were asymptomatic by that time point. It was also chosen because residual subacute thrombus beyond 6 weeks would likely be wall adherent and pose minimal risk of thromboembolism while anticoagulation was discontinued perioperatively.
Our post-treatment imaging included only upper extremity venous duplex examinations. Although 60% of postoperative duplex scans demonstrated fully patent axillosubclavian veins, 40% demonstrated wall thickening and recanalization. As previous authors have documented the limitations of ultrasound examination in detecting nonocclusive mural thrombus in the subclavian vein,19 the authors acknowledge that a greater number of venous abnormalities may have been documented had we consistently performed post-therapy venograms.
Although relying on only post-treatment venous duplex examinations may have underestimated the presence of venous abnormalities, clinical outcomes remained satisfactory over the short and medium term. Both Machleder and Guzzo et al reported clinical outcomes consistently more favorable than postoperative venography might suggest. This current study likewise suggests that eliminating routine preoperative thrombolysis and postoperative venography maintains favorable clinical outcomes, while substantially decreasing overall treatment costs and associated procedural risks.
In 19 of the 20 FRRS cases reported here, the supraclavicular approach was used. Through the supraclavicular exposure, the first rib can be resected from just beyond the costocervical joint to just anterior to the clavicle to insure removal of the rib's anteromedial aspect. Before 2012, patients with PSS at our hospital often underwent post-FRRS venograms that demonstrated effective subclavian vein decompression following supraclavicular FRRS.
The supraclavicular approach had been used effectively by Reilly and Stoney20 in a blended series of patients without TOS and patients with PSS. More recently, Broussard and Donahue21 have advocated for the use of the supraclavicular approach for all three TOS variants. They cited many reasons, including the excellent exposure of the entire first rib from the costochondral cartilage to the head of the rib posteriorly, facilitating scalene excision and vascular reconstruction as necessary.
Several authors have advocated for alternative surgical exposures for venous TOS decompression. Many prefer the transaxillary approach for its less constrained exposure of the anterior first rib, subclavius muscle, and costoclavicular ligament.4,12 Others prefer the paraclavicular approach, which combines both the full neurovascular exposure of the supraclavicular incision with the less obscured view of the costoclavicular joint afforded by the infraclavicular exposure.22 Madden et al6 recently reported on their results transitioning from the paraclavicular to solely infraclavicular approach. They concluded that the infraclavicular approach combined with adjunctive balloon angioplasty was safe, with shorter operative times, and less technical challenge.6 Most recently, Bozzay et al23 reported fewer postoperative symptoms and improved subclavian venous patency in a small group of nonrandomized patients undergoing either infraclavicular vs supraclavicular FRRS for VTOS. Pending a randomized trial comparing approaches, the absolute advantage of one approach remains to be determined.
This clinical patient series has several study limitations. The study group is small and warrants extension to a larger cohort. The authors acknowledge that this study's small cohort size lends itself to possible sampling error. The follow-up, although longer than many previous reports, is nonetheless short among a group of such young patients. Additionally, imaging follow-up is often limited to one venous duplex scan performed at the completion of therapy. Much of the medium-term follow-up (beyond 12 months) during the coronavirus disease 2019 era has been virtual and, therefore, has more limited physical examination capabilities. Occult recurrence is potentially missed.
None of the patients in this series presented with limb-threatening phlegmasia. Rather, they typically presented with moderate arm swelling and cyanosis. The applicability of this simplified algorithm to the most severe clinical presentations of PSS remains in question.
The current study incorporated patients treated with varied anticoagulant regimens, reflecting a decade of progressive transition from warfarin to novel oral anticoagulants. Although these agents are currently used interchangeably, it is possible that one is superior.
With these limitations noted, we here describe a limited series of acute patients with PSS treated with anticoagulation at presentation, an interval delayed first rib resection, and then completion of anticoagulation course. Reflecting the last decade of literature, the treatment algorithm is straightforward, cost saving, and effective through the short to medium term. Clinical outcomes suggest that patients acutely presenting with PSS with moderate symptoms can be treated effectively with anticoagulation and thoracic outlet decompression without thrombolysis. The long-term effectiveness of this treatment on a larger sample size is yet to be demonstrated; hence, this treatment algorithm should be applied with caution. A randomized trial comparing patients treated with and without preoperative thrombolysis is warranted.
Conclusions
Substituting anticoagulation alone for thrombolytic therapy before first rib resection for patients acutely presenting with venous TOS resulted in adequate axillosubclavian venous patency and excellent clinical outcomes though short- and medium-term follow-up.
Author Contributions
Conception and design: ML
Analysis and interpretation: PM, MM, FA, DN, RL, ML
Data collection: PM, MM, ML
Writing the article: PM, ML
Critical revision of the article: PM, MM, FA, DN, RL, ML
Final approval of the article: PM, MM, FA, DN, RL, ML
Statistical analysis: Not applicable
Obtained funding: Not applicable
Overall responsibility: ML
Disclosures
None.
From the Society for Clinical Vascular Surgery
Footnotes
The editors and reviewers of this article have no relevant financial relationships to disclose per the Journal policy that requires reviewers to decline review of any manuscript for which they may have a conflict of interest.
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