- Email: [email protected]
Internal jugular deep venous thrombosis after surgical treatment of a humeral nonunion: a case report and review of the literature
CASE REPORTS Internal jugular deep venous thrombosis after surgical treatment of a humeral nonunion: A case report and review of the literature Albert W. Pearsall IV, MD, David A. Stokes, MD, and George V. Russell Jr, MD, Mobile, AL
U
pper extremity deep venous thrombosis (DVT) is a rare entity comprising approximately 1% to 2% of all cases of venous thromboses. Once thought to be a benign process, DVT in the axillary-subclavian system can lead to prolonged pain and swelling in many patients.13 Classically, upper extremity DVT is divided into primary or effort thrombosis (Paget-Schroetter syndrome) and secondary thrombosis. Primary causes are those inherent to the anatomic structures in the thoracic outlet or axillary region. Secondary causes include trauma, infection, neoplasms, central venous catheters, and hypercoagulable states. Whereas primary thrombosis usually occurs in young active individuals, secondary thrombosis can occur from any of the etiologic factors described above. Although subclavian and axillary vein thrombosis has been reported, we are unaware of a reported case of an internal jugular vein thrombosis after upper extremity fracture surgery.
CASE REPORT A 52-year-old man presented to our outpatient clinic 3 years after intramedullary nail placement for a left humeral shaft fracture. On examination, the patient had pain and motion at the fracture site. Radiographs confirmed a nonunion of the fracture (Figure 1). He subsequently underwent uneventful removal of his intramedullary rod, rotator cuff repair, and open reduction–internal fixation of the humeral nonunion with a 4.5-mm low-contact dynamic compression plate and a 2.7-mm compression plate (Synthes, Paoli, PA) placed 90° to each other around the circumference of the From the Department of Orthopaedic Surgery, University of South Alabama. Reprint requests: Albert W. Pearsall IV, MD, Director, Division of Sports Medicine & Shoulder Service, Department of Orthopaedic Surgery, University of South Alabama, 2451 Fillingim St, MSN 518, Mobile, AL 36617-2293. J Shoulder Elbow Surg 2004;13:459 – 62. Copyright © 2004 by Journal of Shoulder and Elbow Surgery Board of Trustees. 1058-2746/2004/$30.00 doi:10.1016/j.jse.2004.01.015
shaft of the humerus. At the time of surgery, the surgeons considered this construct to be necessary to achieve adequate compression and fixation of the nonunion. Autologous iliac crest bone grafting was also performed at the time of open reduction–internal fixation (Figure 2). On review of the operative procedure, no abnormal maneuvers, difficulty in hardware removal, or prolonged operative period was encountered. The patient was discharged from the hospital on the following day. On the fifth postoperative day, he reported to the local emergency department complaining of acute pain and swelling in his left arm. An upper extremity ultrasonogram revealed a DVT in the left internal jugular vein at the junction of the subclavian and internal jugular veins (Figure 3). He was immediately given intravenous heparin and oral warfarin per recommendations by an internal medicine consultation. During the next 24 hours, the patient’s pain and swelling gradually but completely resolved. He was subsequently discharged from the hospital when his international normalized ratio (INR) was stable at 2.5 IU on oral warfarin. He continued to take warfarin for 3 months with clotting studies checked regularly. A repeat duplex ultrasonogram at 3 months after surgery demonstrated complete restoration of blood flow at the junction of the internal jugular and subclavian veins, the site of the previous thrombosis (Figure 4). At 24 months’ follow-up, the patient was pain-free, had bridging callous radiographically at the fracture site, had no upper extremity edema, and was otherwise symptom-free regarding his DVT.
DISCUSSION DVT of the upper extremity is uncommon.13 Paget and von Schroetter independently described subclavian-axillary vein thrombosis in the latter part of the nineteenth century. Hughes9 later reviewed 320 cases of spontaneous upper limb venous thrombosis and named this condition PagetSchroetter syndrome. Although many descriptions1 have been inconsistently used to describe Paget-Schroetter syndrome, several etiologic factors appear to predispose an individual to the syndrome: strenuous upper body activity, the presence of cervical ribs, congenital bands, scarring, local tissue damage, systemic disease, local intravascular trauma, and oral contraceptive use. Clinical symptoms of upper extremity DVT classically include peripheral edema, prominent superficial veins, and
459
460
Pearsall, Stokes, and Russell
J Shoulder Elbow Surg July/August 2004
Figure 1 Anteroposterior (A) and lateral (B) radiographs of humeral nonunion.
Figure 2 Postoperative anteroposterior (A) and lateral (B) radiographs of humerus after open reduction–internal fixation and iliac crest bone grafting.
J Shoulder Elbow Surg Volume 13, Number 4
Figure 3 Duplex ultrasonogram on postoperative day 5 showing DVT at the junction of the internal jugular vein (IJV) and subclavian vein (SUBCL).
Figure 4 Repeat duplex ultrasonogram of the junction of the internal jugular vein (IJV) and subclavian vein (SUBCL) at 3 months showing return of normal flow and resolution of DVT.
discoloration. Neurologic symptoms may also be present and include pain, paraesthesias, and motor weakness.9 Our patient had all of these clinical manifestations, which enabled an early diagnosis and subsequent treatment. The use of Doppler ultrasound has been shown to be safe and reliable in the diagnosis of subclavian-axillary DVT.11 Several authors have emphasized its accuracy in monitoring patients with indwelling subclavian catheters for clot development. It has been noted that the use of duplex ultrasound does have limitations in the form of acoustic shadowing by the clavicle, which can cause a blind spot in the subclavian vein. In addition, large collaterals can de-
Pearsall, Stokes, and Russell
461
velop, making the interpretation of the scan difficult, leading to a large number of false-negative results.6 Haire et al6 have suggested that although a positive ultrasound can preclude the need for venography, when clinical symptoms are present in the face of a negative Doppler scan, venography is required. Our patient had both classic clinical signs and a positive Doppler ultrasound that led to the diagnosis of internal jugular DVT. The injury mechanism in our case was puzzling, as our patient had normal clotting times recorded preoperatively, and no central or peripheral venous catheter was placed on the affected side in the perioperative period. No additional clotting abnormality studies were performed. The patient did have a 30 –pack-year smoking history that may have predisposed him to venous thrombosis. Smoking has been shown to damage vascular endothelium, promote vascular thrombosis, and increase the relative risk of venous thromboembolism. Hansson et al7 reported that smoking more than 15 cigarettes per day can increase the relative risk 2-fold over that of age-matched nonsmoking control subjects.7 In addition to the patient’s smoking history, the other possible factor contributing to his thrombosis may have been humeral external rotation and traction that were required during surgery. Hyperabduction or prolonged stretching of the affected extremity may damage the intimal wall of the axillary or subclavian vein. Although such maneuvers are frequently used without incident during shoulder surgery, in our patient, such a maneuver may have been sufficient to lead to thrombosis. Cervical ribs have also been associated with upper extremity DVT; however, a postoperative radiographic cervical spine series demonstrated no such abnormality.12 It should be noted that the majority of mechanisms proposed in our discussion are hypothetical, as no distinct event appears to have heralded the onset of our patient’s dramatic symptoms. Indeed, he was discharged without symptoms the day after surgery and did not report any to the hospital or clinical staff until he presented to the emergency department on the fifth postoperative day. Reports have noted long-term sequelae after upper extremity DVT, including chronic pain and edema.14 In addition, the incidence of pulmonary embolism after upper extremity DVT is approximately 14%.3 Original treatment prescriptions for upper extremity DVT included bed rest and elevation of the affected extremity.10 Recently, however, various authors have reported more aggressive treatment options including anticoagulation, antithrombolytics, surgical decompression of anatomic abnormality,12 fibrinolytic therapy,4 and venous dilatation to avoid prolonged symptoms. Anticoagulation has been shown to decrease the acute and chronic morbidity rates significantly compared with conservative treatment alone in upper extremity DVT5 and has been recommended as initial therapy for all upper extremity DVT cases.2 Anticoagulation helps prevent clot propagation in venous collaterals and protects against pulmonary embolism. Hicken and Ameli8 used an algorithm that we used during our treatment process that takes numerous factors into consideration. Given our patient’s rapid response to anticoagulation, further invasive procedures were not undertaken. In our patient, early arm elevation, warm compresses, heparinization, and 3 months of warfarin therapy were used. Currently, he is without symptoms at
462
Pearsall, Stokes, and Russell
24 months after surgery with venous ultrasound documentation of resolution of the thrombosis.
Conclusions Although rare, the clinician should be alert for upper extremity symptoms of swelling, pain, and/or discoloration in order to diagnose upper extremity DVT promptly and correctly. Treatment should include prompt anticoagulation or fibrinolytic therapy, followed by 3 months of continued anticoagulation. Although our patient is currently asymptomatic, patients should be advised of potential long-term sequelae. With the well-documented decrease in thromboembolism seen in lower extremity surgery (ie, total joint arthroplasty) with anticoagulation, perhaps anticoagulation protocols should be examined for revision surgery of the upper extremity as well. REFERENCES
1. Aziz S, Straehley C, Whelan T. Effort-related axillosubclavian vein thrombosis. A new theory of pathogenesis and a plea for direct surgical intervention. Am J Surg 1986;152:57-61. 2. Becker D, Philbrick J, Walker F. Axillary and subclavian venous thrombosis: prognosis and treatment. Arch Intern Med 1991; 151:1934-43. 3. Black M, French G, Rasuli P, Bouchard A. Upper extremity deep venous thrombosis. Underdiagnosed and potentially lethal. Chest 1993;103:1887-90.
J Shoulder Elbow Surg July/August 2004
4. Druy E, Trout H, Giordano J, Hix W. Lytic therapy in the treatment of axillary and subclavian vein thrombosis. J Vasc Surg 1985;2: 821-7. 5. Gloviczki P, Kazmier F, Hollier L. Axillary-subclavian venous occlusion: the morbidity of a nonlethal disease. J Vasc Surg 1986;4:333-7. 6. Haire W, Lynch T, Lieberman R, Lund G, Edney J. Utility of duplex ultrasound in the diagnosis of asymptomatic catheterinduced subclavian vein thrombosis. J Ultrasound Med 1991; 10:493-6. 7. Hansson P, Eriksson H, Welin L, Svardsudd K, Wilhelmsen L. Smoking and abdominal obesity: risk factors for venous thromboembolism among middle-aged men: “the study of men born in 1913.” Arch Intern Med 1999;159:1886-90. 8. Hicken G, Ameli F. Management of subclavian-axillary vein thrombosis: a review. Can J Surg 1998;41:13-25. 9. Hughes E. Venous obstruction in the upper extremity: review of 320 cases. Int Abstr Surg 1949;88:89-127. 10. Inahara T. Surgical treatment of “effort” thrombosis of the axillary and subclavian veins. Am Surg 1968;34:479-83. 11. Kerr T, Lutter K, Moeller D, et al. Upper extremity venous thrombosis diagnosed by duplex scanning. Am J Surg 1990;160: 202-6. 12. Machleder H. Evaluation of a new treatment strategy for PagetSchroetter syndrome: spontaneous thrombosis of the axillarysubclavian vein. J Vasc Surg 1993;17:305-17. 13. Swinton N, Edgett J, Hall R. Primary subclavian axillary vein thrombosis. Circulation 1968;38:737-45. 14. Tilney N, Griffiths H, Edwards E. Natural history of major venous thrombosis of the upper extremity. Arch Surg 1970; 101:792-6.
U
pper extremity deep venous thrombosis (DVT) is a rare entity comprising approximately 1% to 2% of all cases of venous thromboses. Once thought to be a benign process, DVT in the axillary-subclavian system can lead to prolonged pain and swelling in many patients.13 Classically, upper extremity DVT is divided into primary or effort thrombosis (Paget-Schroetter syndrome) and secondary thrombosis. Primary causes are those inherent to the anatomic structures in the thoracic outlet or axillary region. Secondary causes include trauma, infection, neoplasms, central venous catheters, and hypercoagulable states. Whereas primary thrombosis usually occurs in young active individuals, secondary thrombosis can occur from any of the etiologic factors described above. Although subclavian and axillary vein thrombosis has been reported, we are unaware of a reported case of an internal jugular vein thrombosis after upper extremity fracture surgery.
CASE REPORT A 52-year-old man presented to our outpatient clinic 3 years after intramedullary nail placement for a left humeral shaft fracture. On examination, the patient had pain and motion at the fracture site. Radiographs confirmed a nonunion of the fracture (Figure 1). He subsequently underwent uneventful removal of his intramedullary rod, rotator cuff repair, and open reduction–internal fixation of the humeral nonunion with a 4.5-mm low-contact dynamic compression plate and a 2.7-mm compression plate (Synthes, Paoli, PA) placed 90° to each other around the circumference of the From the Department of Orthopaedic Surgery, University of South Alabama. Reprint requests: Albert W. Pearsall IV, MD, Director, Division of Sports Medicine & Shoulder Service, Department of Orthopaedic Surgery, University of South Alabama, 2451 Fillingim St, MSN 518, Mobile, AL 36617-2293. J Shoulder Elbow Surg 2004;13:459 – 62. Copyright © 2004 by Journal of Shoulder and Elbow Surgery Board of Trustees. 1058-2746/2004/$30.00 doi:10.1016/j.jse.2004.01.015
shaft of the humerus. At the time of surgery, the surgeons considered this construct to be necessary to achieve adequate compression and fixation of the nonunion. Autologous iliac crest bone grafting was also performed at the time of open reduction–internal fixation (Figure 2). On review of the operative procedure, no abnormal maneuvers, difficulty in hardware removal, or prolonged operative period was encountered. The patient was discharged from the hospital on the following day. On the fifth postoperative day, he reported to the local emergency department complaining of acute pain and swelling in his left arm. An upper extremity ultrasonogram revealed a DVT in the left internal jugular vein at the junction of the subclavian and internal jugular veins (Figure 3). He was immediately given intravenous heparin and oral warfarin per recommendations by an internal medicine consultation. During the next 24 hours, the patient’s pain and swelling gradually but completely resolved. He was subsequently discharged from the hospital when his international normalized ratio (INR) was stable at 2.5 IU on oral warfarin. He continued to take warfarin for 3 months with clotting studies checked regularly. A repeat duplex ultrasonogram at 3 months after surgery demonstrated complete restoration of blood flow at the junction of the internal jugular and subclavian veins, the site of the previous thrombosis (Figure 4). At 24 months’ follow-up, the patient was pain-free, had bridging callous radiographically at the fracture site, had no upper extremity edema, and was otherwise symptom-free regarding his DVT.
DISCUSSION DVT of the upper extremity is uncommon.13 Paget and von Schroetter independently described subclavian-axillary vein thrombosis in the latter part of the nineteenth century. Hughes9 later reviewed 320 cases of spontaneous upper limb venous thrombosis and named this condition PagetSchroetter syndrome. Although many descriptions1 have been inconsistently used to describe Paget-Schroetter syndrome, several etiologic factors appear to predispose an individual to the syndrome: strenuous upper body activity, the presence of cervical ribs, congenital bands, scarring, local tissue damage, systemic disease, local intravascular trauma, and oral contraceptive use. Clinical symptoms of upper extremity DVT classically include peripheral edema, prominent superficial veins, and
459
460
Pearsall, Stokes, and Russell
J Shoulder Elbow Surg July/August 2004
Figure 1 Anteroposterior (A) and lateral (B) radiographs of humeral nonunion.
Figure 2 Postoperative anteroposterior (A) and lateral (B) radiographs of humerus after open reduction–internal fixation and iliac crest bone grafting.
J Shoulder Elbow Surg Volume 13, Number 4
Figure 3 Duplex ultrasonogram on postoperative day 5 showing DVT at the junction of the internal jugular vein (IJV) and subclavian vein (SUBCL).
Figure 4 Repeat duplex ultrasonogram of the junction of the internal jugular vein (IJV) and subclavian vein (SUBCL) at 3 months showing return of normal flow and resolution of DVT.
discoloration. Neurologic symptoms may also be present and include pain, paraesthesias, and motor weakness.9 Our patient had all of these clinical manifestations, which enabled an early diagnosis and subsequent treatment. The use of Doppler ultrasound has been shown to be safe and reliable in the diagnosis of subclavian-axillary DVT.11 Several authors have emphasized its accuracy in monitoring patients with indwelling subclavian catheters for clot development. It has been noted that the use of duplex ultrasound does have limitations in the form of acoustic shadowing by the clavicle, which can cause a blind spot in the subclavian vein. In addition, large collaterals can de-
Pearsall, Stokes, and Russell
461
velop, making the interpretation of the scan difficult, leading to a large number of false-negative results.6 Haire et al6 have suggested that although a positive ultrasound can preclude the need for venography, when clinical symptoms are present in the face of a negative Doppler scan, venography is required. Our patient had both classic clinical signs and a positive Doppler ultrasound that led to the diagnosis of internal jugular DVT. The injury mechanism in our case was puzzling, as our patient had normal clotting times recorded preoperatively, and no central or peripheral venous catheter was placed on the affected side in the perioperative period. No additional clotting abnormality studies were performed. The patient did have a 30 –pack-year smoking history that may have predisposed him to venous thrombosis. Smoking has been shown to damage vascular endothelium, promote vascular thrombosis, and increase the relative risk of venous thromboembolism. Hansson et al7 reported that smoking more than 15 cigarettes per day can increase the relative risk 2-fold over that of age-matched nonsmoking control subjects.7 In addition to the patient’s smoking history, the other possible factor contributing to his thrombosis may have been humeral external rotation and traction that were required during surgery. Hyperabduction or prolonged stretching of the affected extremity may damage the intimal wall of the axillary or subclavian vein. Although such maneuvers are frequently used without incident during shoulder surgery, in our patient, such a maneuver may have been sufficient to lead to thrombosis. Cervical ribs have also been associated with upper extremity DVT; however, a postoperative radiographic cervical spine series demonstrated no such abnormality.12 It should be noted that the majority of mechanisms proposed in our discussion are hypothetical, as no distinct event appears to have heralded the onset of our patient’s dramatic symptoms. Indeed, he was discharged without symptoms the day after surgery and did not report any to the hospital or clinical staff until he presented to the emergency department on the fifth postoperative day. Reports have noted long-term sequelae after upper extremity DVT, including chronic pain and edema.14 In addition, the incidence of pulmonary embolism after upper extremity DVT is approximately 14%.3 Original treatment prescriptions for upper extremity DVT included bed rest and elevation of the affected extremity.10 Recently, however, various authors have reported more aggressive treatment options including anticoagulation, antithrombolytics, surgical decompression of anatomic abnormality,12 fibrinolytic therapy,4 and venous dilatation to avoid prolonged symptoms. Anticoagulation has been shown to decrease the acute and chronic morbidity rates significantly compared with conservative treatment alone in upper extremity DVT5 and has been recommended as initial therapy for all upper extremity DVT cases.2 Anticoagulation helps prevent clot propagation in venous collaterals and protects against pulmonary embolism. Hicken and Ameli8 used an algorithm that we used during our treatment process that takes numerous factors into consideration. Given our patient’s rapid response to anticoagulation, further invasive procedures were not undertaken. In our patient, early arm elevation, warm compresses, heparinization, and 3 months of warfarin therapy were used. Currently, he is without symptoms at
462
Pearsall, Stokes, and Russell
24 months after surgery with venous ultrasound documentation of resolution of the thrombosis.
Conclusions Although rare, the clinician should be alert for upper extremity symptoms of swelling, pain, and/or discoloration in order to diagnose upper extremity DVT promptly and correctly. Treatment should include prompt anticoagulation or fibrinolytic therapy, followed by 3 months of continued anticoagulation. Although our patient is currently asymptomatic, patients should be advised of potential long-term sequelae. With the well-documented decrease in thromboembolism seen in lower extremity surgery (ie, total joint arthroplasty) with anticoagulation, perhaps anticoagulation protocols should be examined for revision surgery of the upper extremity as well. REFERENCES
1. Aziz S, Straehley C, Whelan T. Effort-related axillosubclavian vein thrombosis. A new theory of pathogenesis and a plea for direct surgical intervention. Am J Surg 1986;152:57-61. 2. Becker D, Philbrick J, Walker F. Axillary and subclavian venous thrombosis: prognosis and treatment. Arch Intern Med 1991; 151:1934-43. 3. Black M, French G, Rasuli P, Bouchard A. Upper extremity deep venous thrombosis. Underdiagnosed and potentially lethal. Chest 1993;103:1887-90.
J Shoulder Elbow Surg July/August 2004
4. Druy E, Trout H, Giordano J, Hix W. Lytic therapy in the treatment of axillary and subclavian vein thrombosis. J Vasc Surg 1985;2: 821-7. 5. Gloviczki P, Kazmier F, Hollier L. Axillary-subclavian venous occlusion: the morbidity of a nonlethal disease. J Vasc Surg 1986;4:333-7. 6. Haire W, Lynch T, Lieberman R, Lund G, Edney J. Utility of duplex ultrasound in the diagnosis of asymptomatic catheterinduced subclavian vein thrombosis. J Ultrasound Med 1991; 10:493-6. 7. Hansson P, Eriksson H, Welin L, Svardsudd K, Wilhelmsen L. Smoking and abdominal obesity: risk factors for venous thromboembolism among middle-aged men: “the study of men born in 1913.” Arch Intern Med 1999;159:1886-90. 8. Hicken G, Ameli F. Management of subclavian-axillary vein thrombosis: a review. Can J Surg 1998;41:13-25. 9. Hughes E. Venous obstruction in the upper extremity: review of 320 cases. Int Abstr Surg 1949;88:89-127. 10. Inahara T. Surgical treatment of “effort” thrombosis of the axillary and subclavian veins. Am Surg 1968;34:479-83. 11. Kerr T, Lutter K, Moeller D, et al. Upper extremity venous thrombosis diagnosed by duplex scanning. Am J Surg 1990;160: 202-6. 12. Machleder H. Evaluation of a new treatment strategy for PagetSchroetter syndrome: spontaneous thrombosis of the axillarysubclavian vein. J Vasc Surg 1993;17:305-17. 13. Swinton N, Edgett J, Hall R. Primary subclavian axillary vein thrombosis. Circulation 1968;38:737-45. 14. Tilney N, Griffiths H, Edwards E. Natural history of major venous thrombosis of the upper extremity. Arch Surg 1970; 101:792-6.