- Email: [email protected]
Peroneal Nerve Palsy Caused by Popliteal Pseudoaneurysm in A Child with Hereditary Multiple Exostosis
Peroneal Nerve Palsy Caused by Popliteal Pseudoaneurysm in A Child with Hereditary Multiple Exostosis Burak Onan, Ismihan Selen Onan, Yesim Guner, and Mehmet Yeniterzi, Istanbul, Turkey
A 12-year-old boy with a family history of hereditary multiple exostosis presented with a 3-week history of progressive knee swelling. The clinical examination revealed drop foot and a loss of extension in his right knee. Evaluation with color duplex ultrasonography, computed tomography angiography, and magnetic resonance imaging revealed a popliteal artery pseudoaneurysm associated with exostosis from the distal femur. This patient was diagnosed as having peroneal neuropathy caused by popliteal artery pseudoaneurysm compressing the nerve in the right popliteal fossa. The pseudoaneurysm was repaired primarily, and the exostosis was excised during the operation. Pain and knee contracture resolved after surgery. The patient was then referred to physical therapy for the management of drop foot.
Peroneal nerve palsy associated with compression is the most common mononeuropathy of the lower extremity.1,2 The etiology of peroneal nerve palsy frequently includes blunt or penetrating trauma, fibular fracture, metabolic syndromes, ischemic neuropathy, spontaneous hematoma, entrapment or neoplasms.3 However, a pseudoaneurysm in the popliteal fossa compressing the peroneal nerve is an unusual cause for peroneal nerve palsy.3e5 In this report, we describe peroneal nerve palsy due to pseudoaneurysm of the popliteal artery in a child with hereditary multiple exostosis.
Department of Cardiovascular Surgery, Istanbul Mehmet Akif Ersoy Thoracic and Cardiovascular Surgery Education and Training Hospital, Istanbul, Turkey. Correspondence to: Burak Onan, MD, Department of Cardiovascular Surgery, Istanbul Mehmet Akif Ersoy Thoracic and Cardiovascular Surgery, Training and Education Hospital, Istasyon Mahallesi, Turgut Ozal Bulvari, No. 11, 34303 Kucukcekmece, Istanbul, Turkey; E-mail: [email protected] Ann Vasc Surg 2014; 28: 1037.e5–1037.e9 http://dx.doi.org/10.1016/j.avsg.2013.08.018 Ó 2014 Elsevier Inc. All rights reserved. Manuscript received: April 6, 2013; manuscript accepted: August 13, 2013; published online: December 9, 2013.
CASE REPORT A 12 year-old male with a family history of hereditary multiple exostosis presented with progressive pain and swelling in his right popliteal area. The clinical examination revealed drop foot and a loss of extension in his right knee. The swelling and associated symptoms had increased over the course of the previous 3 weeks. The pain was resistant to analgesic drugs, including acetaminophen and nonsteroidal anti-inflammatory drugs. The patient had no fever, weight loss, or history of trauma. The physical examination revealed swelling that was localized to the anteromedial and posterior aspect of the right lower thigh. This area was tender and warm on palpation. There was a motor deficit in the right foot and a marked weakness in extension of the right knee. The patient had a flexion deformity of the knee joint because of swelling in the right popliteal fossa. The distal arteries were all palpable, including the dorsalis pedis and posterior tibialis arteries. There was no edema of the lower extremities that would suggest a venous or lymphatic disease process. Biochemical tests were normal. Doppler ultrasonography with color mode revealed a pseudoaneurysm (105 75 mm) of the right popliteal artery in the popliteal fossa. A computed tomography angiography scan of the right lower extremity revealed a lobulated mass in the popliteal fossa (Fig. 1). The lesion was closely associated with bony spikes from the distal femur (Fig. 1, inset). There were also multiple osteochondromas
1037.e5
1037.e6 Case reports
Annals of Vascular Surgery
consultant ordered an anticonvulsive agent, gabapentin, 100 mg twice a day. When the symptoms improved with medical treatment, gentle stretching and range of motion exercises increased the mobility of the knee joint. The full range of knee motion was achieved in 6 weeks. The patient was then referred to physical therapy for the management of drop foot.
DISCUSSION
Fig. 1. Computed tomography images revealing a pseudoaneurysm of the right popliteal artery. The images also revealed multiple bony spikes from the distal femur (inset; white arrows). PA, Popliteal artery; PPA, pseudoaneurysm of the popliteal artery.
on both of the lower extremities. A magnetic resonance imaging scan confirmed the lesion and revealed the close association of the exocytosis from the distal femur and the pseudoaneurysm sac (Fig. 2). Electrophysiologic studies, including electromyography and nerve conduction studies, suggested peroneal nerve palsy that might be caused by external compression or traction by the pseudoaneurysm sac. Because this patient had a progressively growing lesion that was associated with peroneal neuropathy, a surgical intervention was planned to relieve the compression to the peroneal nerve by the pseudoaneurysm. The pseudoaneurysm sac was explored through an incision over the medial aspect of the right lower thigh. The right common femoral artery and the right popliteal artery below the knee joint were both secured before this incision in order to prevent any intraoperative bleeding. The exploration was first started superiorly to expose the superficial femoral artery, and then the aneurysm sac was entered. The sac was compressing and displacing the popliteal artery posteromedially. After evacuation of the blood and hematoma, multiple bony spikes were palpated in the posteromedial aspect of the distal femur (Fig. 3). These spikes were the potential cause for an injury to the popliteal artery and associated pseudoaneurysm formation. The segment of the popliteal artery that was lacerated was resected and an end-to-end anastomosis with 6e0 polypropylene suture was performed to repair this defect (Fig. 3, inset). A consulting orthopedic surgeon excised the bony spikes from the popliteal fossa. The operation was completed uneventfully. The swelling and associated local findings resolved dramatically postoperatively. Peripheral arterial and venous circulation was normal. The pain did not respond to conventional medications and opioids. A neurology
Peroneal nerve palsy is a rare complication of pseudoaneurysms of the popliteal artery, especially in childhood.3e5 Its clinical diagnosis is suggested with the presence of weakness of the ankle dorsiflexion and eversion and toe extension. The pathogenesis of this rare event frequently includes blunt or penetrating trauma to the lower extremities.6 In such cases, peroneal nerve injury can be associated with traction, laceration, or compression of the nerve caused by bone fracture or hematoma. Specifically, the presence of aneurysm (true or false) formation in the popliteal fossa may also lead to the compression or traction of the peroneal nerve.3e6 In our patient, a pseudoaneurysm in the popliteal area was a predisposing factor for the development of peroneal nerve palsy. The mechanism of peroneal nerve palsy can be clarified with an understanding of the anatomic course of the nerve.6 The largest nerve in the sacral plexus and in the human body is the sciatic nerve. It receives contributions from the L4eS3 spinal nerves. The sciatic nerve divides into the common peroneal and tibial nerves proximally to the popliteal fossa. The common peroneal nerve divides into the superficial peroneal nerve for the lateral leg compartment and the deep peroneal nerve for the deep leg compartment. The branches of the peroneal nerve innervate the short head of the bicep femoris muscle and the tibialis anterior muscle, which is the major contributor to foot dorsiflexion. Other muscles, including the extensor digitorum longus and peroneus tertius, also assist with dorsiflexion of the foot. Therefore, any space-occupying lesion within the popliteal fossa may jeopardize the peroneal nerve and cause associated clinical findings. The common vascular complications of hereditary multiple exostosis in the pediatric population include pseudoaneurysm formation, claudication, acute ischemia, and phlebitis.7e10 The presentation of a popliteal pseudoaneurysm with peroneal palsy is unusual in patients with hereditary multiple exostosis.3e5 In this hereditary disorder, multiple bony spikes can be seen in different locations of the extremities. In our patient, bony spikes from the distal
Vol. 28, No. 4, May 2014
Case reports 1037.e7
Fig. 2. (AeD) Magnetic resonance imaging scan revealing multiple bony spikes from the distal segment of the right femur (white arrows).
Fig. 3. Operative view after evacuation of blood and hematoma from the pseudoaneurysm sac reveals bony spikes from the distal femur (white arrows) and the lesion in the right popliteal artery (black arrows).
femur were diagnosed during preoperative computed tomography and magnetic resonance imaging studies and during surgical exploration. They were
extending in multiple directions in the popliteal fossa. Anatomically, the popliteal artery courses posteriorly to the distal femur in the popliteal fossa. It is fixed proximally at the Hunter canal and distally at the trifurcation. Therefore, both the proximal and distal portions of the popliteal artery have little mobility and are prone to injury by spikes. The constant rubbing against the exostosis during movement can cause continuous damage to the fixed wall of the artery, eventually leading to pseudoaneurysm formation.7e10 During the operation, we observed that the spikes were stiff, sharp, and irregular lesions. They were excised to prevent additional neurovascular injuries. We suggest that clinicians should be aware that potential neurovascular injuries can develop in patients with hereditary multiple exostosis during clinical follow-up. Arterial pseudoaneurysms should be included in the differential diagnosis of potential complications of hereditary multiple exostosis.7e9 If these patients present with a swelling in the popliteal area, they should be immediately referred for a radiologic
1037.e8 Case reports
Annals of Vascular Surgery
Table I. Main etiology, presentation, and management of peroneal nerve palsy and associated pathologies Main etiology
Musculoskeletal injury
1,3,6
Mass lesions11,12
Malnutrition6,13,14 Intermittent pneumatic compression15 Osteoarthritis16 Vascular lesions and injuries3e5 Neurotoxicity17 Systemic disorder18,19
Presentation/cause
Management
Tibia or fibular fracture, knee arthroplasty, orthopedic surgery, penetrating or blunt limb injuries, and compartment syndrome Intraneural ganglion cysts, tumors, such as lipoma, schwannoma, neurofibroma, osteochondroma, neurogenic sarcoma, focal hypertrophic neuropathy, desmoid tumor, or bone tumors, hematoma, and abscess Weight loss in patients with prolonged bed rest Deep venous thrombosis
Treatment of underlying cause, physiotherapy, analgesic agents, neurolysis, or nerve grafting Treatment for mass lesions, decompression of the nerve, and analgesic agents
Physiotherapy and analgesic agents
Treatment for venous thrombosis, elevation, analgesic agents, and anticoagulation Knee deformity and associated nerve stretch Orthopedic management, physiotherapy, and analgesic agents True or false arterial aneurysms or limb Decompression of the mass lesion, vascular injury repair, physiotherapy, and analgesic agents Antineoplastic agents Change of antineoplastic agent Isolated nerve palsy Treatment of systemic disorder and analgesic agents
evaluation. Briefly, a single radiograph and computed tomography scan can help to establish anatomic relationships between exostosis, pseudoaneurysm, and the affected artery. The diagnosis of pseudoaneurysm is generally made by color Duplex ultrasonography. Nevertheless, contrast-enhanced computed tomography and magnetic resonance imaging studies are helpful to define the relationship between the exostosis and pseudoaneurysm of the popliteal artery. A 3-dimensional reconstruction of the images also gives spatial information of the lesions. The management of nerve compression syndromes depends on the etiology11e19 (Table I). The removal of the predisposing cause, lesion, or activity is the best treatment of peroneal neuropathies.6 In pseudoaneurysm of the popliteal artery, it has been noted that the initial treatment of peroneal nerve palsy requires immediate repair of the aneurysm and decompression of the nerve.3e5 If there is no clinical improvement, surgical intervention for neuropathy is then recommended. Neurolysis, nerve grafting, or a combination of the two is considered if there is partial or complete nerve injury, especially in cases of trauma. Garazzo et al.20 reported that common peroneal nerve palsies in open wounds should undergo surgical exploration at emergency. In closed injuries with no spontaneous recovery within 4 months postinjury, patients should be referred for surgical treatment regardless of the causative mechanism of the lesion. In addition, physical
therapy is mandatory to prevent contracture or to relieve deformities caused by a loss of or weakness in the range of motion of the joints. Clinically, pain is considered to be the early symptom in peroneal neuropathy. Its management may be difficult with conventional analgesic agents, such as acetaminophen or nonsteroidal antiinflammatory drugs. Alternative analgesic agents to relieve neuropathic pain, such as antiepileptic drugs, opioids, or selective serotonin uptake inhibitors can be necessary.6 In our patient, gabapentin provided effective postoperative analgesia. Surgical treatment of the vascular complications of exostosis is recommended as an urgent procedure to avoid arterial occlusion, embolism, or phlebitis.10 Pseudoaneurysms can be repaired with primary closure of defect, reconstruction of the artery with a vein patch, resection of the artery and end to end anastomosis, venous grafting, or polytetrafluoroethylene bypass. The pseudoaneurysm sac, including blood and hematoma, is evacuated to decompress the peroneal nerve. With regard to surgical exposure, 3 approaches can be used for popliteal aneurysms: medial, posterior, and lateral. In our case, we used a medial approach to allow for simultaneous control of the common femoral, superficial femoral, and infrapopliteal arteries in the supine position before opening the pseudoaneurysm sac. This approach also enabled exploration of the bony spikes that were closely associated with
Vol. 28, No. 4, May 2014
the popliteal artery. The posterior approach could have been used in this patient, but flexion deformity of the knee joint might have limited adequate exposure of the sac and popliteal artery. In the published literature, there is no clear indication for the timing of excision of the exostosis. Surgical excision has been recommended in painful and enlarging lesions. Some authors recommend surgical repair of the exostosis if it becomes malignant; however, early excision is recommended if the exostosis is next to a major vessel or there is bone fracture or neurovascular injury.10 In our case, after repair of the popliteal artery with an end to end anastomosis, bony spikes were excised. The bony spikes were resected after reconstruction of the artery because there was an enough space in the popliteal fossa after evacuation of the pseudoaneurysm. Nevertheless, in some cases, an intraoperative arterial shunt may be considered to maintain temporary revascularization until the orthopedic portion of the case is completed. In conclusion, peroneal nerve palsy is an unusual complication of popliteal pseudoaneurysms. Patients with hereditary multiple exostoses should be followed-up closely to prevent associated vascular and neurologic injuries. REFERENCES 1. Dawson DM, Hallett M, Wilbourn AJ. Entrapment neuropathies. 3rd ed. Philadelphia: Lippincott-Raven, 1999. 2. McCluskey LF, Webb LB. Compression and entrapment neuropathies of the lower extremity. Clin Podiatr Med Surg 1999;16:97e125. 3. Ersozlu S, Ozulku M, Yildirim E, et al. Common peroneal nerve palsy from an untreated popliteal pseudoaneurysm after penetrating injury. J Vasc Surg 2007;45:408e10. 4. Ozc¸akar L, Aknc A, Aksoy DY, et al. Peroneal neuropathy due to a popliteal aneurysm in a patient with infectious endocarditis. Ann Vasc Surg 2004;18:115e7.
Case reports 1037.e9
5. Megalopoulos A, Vasiliadis K, Siminas S, et al. Pseudoaneurysm of the popliteal artery complicated by peroneal mononeuropathy in a 4-year-old child: report of a case. Surg Today 2007;37:798e801. 6. Baima J, Krivickas L. Evaluation and treatment of peroneal neuropathy. Curr Rev Musculoskelet Med 2008;1:147e53. 7. Pavic P, Vergles D, Sarlija M, et al. Pseudoaneurysm of the popliteal artery in a patient with multiple hereditary exostoses. Ann Vasc Surg 2011;25:268.e1e2. 8. Lieberman J, Mazzucco J, Kwasnik E, et al. Popliteal pseudoaneurysm as a complication of an adjacent osteochondroma. Ann Vasc Surg 1994;8:198e203. 9. Matsushita M, Nishikimi N, Sakurai T, et al. Pseudoaneurysm of the popliteal artery caused by exostosis of the femur: case report and review of the literature. J Vasc Surg 2000;32:201e4. 10. Vasseur MA, Fabre O. Vascular complications of osteochondromas. J Vasc Surg 2000;31:532e8. 11. Kim DH, Murovic JA, Teil RL, et al. Management and outcomes in 318 operative common peroneal nerve lesions at the LSU Health Sciences Center. Neurosurgery 2004;54:1421e9. 12. Kim JY, Ihn YK, Kim JS, et al. Nontraumatic peroneal nerve palsy: MRI findings. Clin Radiol 2007;62:58e64. 13. Sotaniemi KA. Slimmer’s paralysisdperoneal neuropathy during weight reduction. J Neurol Neurosurg Psychiatry 1984;47:564e6. 14. Katirji MB, Wilbourn AJ. Common peroneal mononeuropathy: a clinical and electrophysiologic study of 116 lesions. Neurology 1988;38:1723e8. 15. Fukuda H. Bilateral peroneal nerve palsy caused by intermittent pneumatic compression. Intern Med 2006;45:93e4. 16. Fetzer B, Prather H, Gelberman RH, et al. Progressive peroneal nerve palsy in a varus arthritic knee. A case report. J Bone Joint Surg Am 2004;86:1538e40. 17. Argyriu AA, Bruna J, Marmiroli P, et al. Chemotherapyinduced peripheral neurotoxicity (CIPN): an update. Crit Rev Oncol Hematol 2012;82:51e77. 18. Wolf J, Schmitt V, Palm F, et al. Peripheral neuropathy as initial manifestation of primary systemic vasculitides. J Neurol 2013;260:1061e70. 19. Florica B, Aghdassi E, Su J, et al. Peripheral neuropathy in patients with systemic lupus erythematosus. Semin Arthritis Rheum 2011;41:203e11. 20. Garozzo D, Ferraresi S, Buffatti P. Surgical treatment of common peroneal nerve injuries: indications and results. A series of 62 cases. J Neurosurg Sci 2004;48:105e12.
A 12-year-old boy with a family history of hereditary multiple exostosis presented with a 3-week history of progressive knee swelling. The clinical examination revealed drop foot and a loss of extension in his right knee. Evaluation with color duplex ultrasonography, computed tomography angiography, and magnetic resonance imaging revealed a popliteal artery pseudoaneurysm associated with exostosis from the distal femur. This patient was diagnosed as having peroneal neuropathy caused by popliteal artery pseudoaneurysm compressing the nerve in the right popliteal fossa. The pseudoaneurysm was repaired primarily, and the exostosis was excised during the operation. Pain and knee contracture resolved after surgery. The patient was then referred to physical therapy for the management of drop foot.
Peroneal nerve palsy associated with compression is the most common mononeuropathy of the lower extremity.1,2 The etiology of peroneal nerve palsy frequently includes blunt or penetrating trauma, fibular fracture, metabolic syndromes, ischemic neuropathy, spontaneous hematoma, entrapment or neoplasms.3 However, a pseudoaneurysm in the popliteal fossa compressing the peroneal nerve is an unusual cause for peroneal nerve palsy.3e5 In this report, we describe peroneal nerve palsy due to pseudoaneurysm of the popliteal artery in a child with hereditary multiple exostosis.
Department of Cardiovascular Surgery, Istanbul Mehmet Akif Ersoy Thoracic and Cardiovascular Surgery Education and Training Hospital, Istanbul, Turkey. Correspondence to: Burak Onan, MD, Department of Cardiovascular Surgery, Istanbul Mehmet Akif Ersoy Thoracic and Cardiovascular Surgery, Training and Education Hospital, Istasyon Mahallesi, Turgut Ozal Bulvari, No. 11, 34303 Kucukcekmece, Istanbul, Turkey; E-mail: [email protected] Ann Vasc Surg 2014; 28: 1037.e5–1037.e9 http://dx.doi.org/10.1016/j.avsg.2013.08.018 Ó 2014 Elsevier Inc. All rights reserved. Manuscript received: April 6, 2013; manuscript accepted: August 13, 2013; published online: December 9, 2013.
CASE REPORT A 12 year-old male with a family history of hereditary multiple exostosis presented with progressive pain and swelling in his right popliteal area. The clinical examination revealed drop foot and a loss of extension in his right knee. The swelling and associated symptoms had increased over the course of the previous 3 weeks. The pain was resistant to analgesic drugs, including acetaminophen and nonsteroidal anti-inflammatory drugs. The patient had no fever, weight loss, or history of trauma. The physical examination revealed swelling that was localized to the anteromedial and posterior aspect of the right lower thigh. This area was tender and warm on palpation. There was a motor deficit in the right foot and a marked weakness in extension of the right knee. The patient had a flexion deformity of the knee joint because of swelling in the right popliteal fossa. The distal arteries were all palpable, including the dorsalis pedis and posterior tibialis arteries. There was no edema of the lower extremities that would suggest a venous or lymphatic disease process. Biochemical tests were normal. Doppler ultrasonography with color mode revealed a pseudoaneurysm (105 75 mm) of the right popliteal artery in the popliteal fossa. A computed tomography angiography scan of the right lower extremity revealed a lobulated mass in the popliteal fossa (Fig. 1). The lesion was closely associated with bony spikes from the distal femur (Fig. 1, inset). There were also multiple osteochondromas
1037.e5
1037.e6 Case reports
Annals of Vascular Surgery
consultant ordered an anticonvulsive agent, gabapentin, 100 mg twice a day. When the symptoms improved with medical treatment, gentle stretching and range of motion exercises increased the mobility of the knee joint. The full range of knee motion was achieved in 6 weeks. The patient was then referred to physical therapy for the management of drop foot.
DISCUSSION
Fig. 1. Computed tomography images revealing a pseudoaneurysm of the right popliteal artery. The images also revealed multiple bony spikes from the distal femur (inset; white arrows). PA, Popliteal artery; PPA, pseudoaneurysm of the popliteal artery.
on both of the lower extremities. A magnetic resonance imaging scan confirmed the lesion and revealed the close association of the exocytosis from the distal femur and the pseudoaneurysm sac (Fig. 2). Electrophysiologic studies, including electromyography and nerve conduction studies, suggested peroneal nerve palsy that might be caused by external compression or traction by the pseudoaneurysm sac. Because this patient had a progressively growing lesion that was associated with peroneal neuropathy, a surgical intervention was planned to relieve the compression to the peroneal nerve by the pseudoaneurysm. The pseudoaneurysm sac was explored through an incision over the medial aspect of the right lower thigh. The right common femoral artery and the right popliteal artery below the knee joint were both secured before this incision in order to prevent any intraoperative bleeding. The exploration was first started superiorly to expose the superficial femoral artery, and then the aneurysm sac was entered. The sac was compressing and displacing the popliteal artery posteromedially. After evacuation of the blood and hematoma, multiple bony spikes were palpated in the posteromedial aspect of the distal femur (Fig. 3). These spikes were the potential cause for an injury to the popliteal artery and associated pseudoaneurysm formation. The segment of the popliteal artery that was lacerated was resected and an end-to-end anastomosis with 6e0 polypropylene suture was performed to repair this defect (Fig. 3, inset). A consulting orthopedic surgeon excised the bony spikes from the popliteal fossa. The operation was completed uneventfully. The swelling and associated local findings resolved dramatically postoperatively. Peripheral arterial and venous circulation was normal. The pain did not respond to conventional medications and opioids. A neurology
Peroneal nerve palsy is a rare complication of pseudoaneurysms of the popliteal artery, especially in childhood.3e5 Its clinical diagnosis is suggested with the presence of weakness of the ankle dorsiflexion and eversion and toe extension. The pathogenesis of this rare event frequently includes blunt or penetrating trauma to the lower extremities.6 In such cases, peroneal nerve injury can be associated with traction, laceration, or compression of the nerve caused by bone fracture or hematoma. Specifically, the presence of aneurysm (true or false) formation in the popliteal fossa may also lead to the compression or traction of the peroneal nerve.3e6 In our patient, a pseudoaneurysm in the popliteal area was a predisposing factor for the development of peroneal nerve palsy. The mechanism of peroneal nerve palsy can be clarified with an understanding of the anatomic course of the nerve.6 The largest nerve in the sacral plexus and in the human body is the sciatic nerve. It receives contributions from the L4eS3 spinal nerves. The sciatic nerve divides into the common peroneal and tibial nerves proximally to the popliteal fossa. The common peroneal nerve divides into the superficial peroneal nerve for the lateral leg compartment and the deep peroneal nerve for the deep leg compartment. The branches of the peroneal nerve innervate the short head of the bicep femoris muscle and the tibialis anterior muscle, which is the major contributor to foot dorsiflexion. Other muscles, including the extensor digitorum longus and peroneus tertius, also assist with dorsiflexion of the foot. Therefore, any space-occupying lesion within the popliteal fossa may jeopardize the peroneal nerve and cause associated clinical findings. The common vascular complications of hereditary multiple exostosis in the pediatric population include pseudoaneurysm formation, claudication, acute ischemia, and phlebitis.7e10 The presentation of a popliteal pseudoaneurysm with peroneal palsy is unusual in patients with hereditary multiple exostosis.3e5 In this hereditary disorder, multiple bony spikes can be seen in different locations of the extremities. In our patient, bony spikes from the distal
Vol. 28, No. 4, May 2014
Case reports 1037.e7
Fig. 2. (AeD) Magnetic resonance imaging scan revealing multiple bony spikes from the distal segment of the right femur (white arrows).
Fig. 3. Operative view after evacuation of blood and hematoma from the pseudoaneurysm sac reveals bony spikes from the distal femur (white arrows) and the lesion in the right popliteal artery (black arrows).
femur were diagnosed during preoperative computed tomography and magnetic resonance imaging studies and during surgical exploration. They were
extending in multiple directions in the popliteal fossa. Anatomically, the popliteal artery courses posteriorly to the distal femur in the popliteal fossa. It is fixed proximally at the Hunter canal and distally at the trifurcation. Therefore, both the proximal and distal portions of the popliteal artery have little mobility and are prone to injury by spikes. The constant rubbing against the exostosis during movement can cause continuous damage to the fixed wall of the artery, eventually leading to pseudoaneurysm formation.7e10 During the operation, we observed that the spikes were stiff, sharp, and irregular lesions. They were excised to prevent additional neurovascular injuries. We suggest that clinicians should be aware that potential neurovascular injuries can develop in patients with hereditary multiple exostosis during clinical follow-up. Arterial pseudoaneurysms should be included in the differential diagnosis of potential complications of hereditary multiple exostosis.7e9 If these patients present with a swelling in the popliteal area, they should be immediately referred for a radiologic
1037.e8 Case reports
Annals of Vascular Surgery
Table I. Main etiology, presentation, and management of peroneal nerve palsy and associated pathologies Main etiology
Musculoskeletal injury
1,3,6
Mass lesions11,12
Malnutrition6,13,14 Intermittent pneumatic compression15 Osteoarthritis16 Vascular lesions and injuries3e5 Neurotoxicity17 Systemic disorder18,19
Presentation/cause
Management
Tibia or fibular fracture, knee arthroplasty, orthopedic surgery, penetrating or blunt limb injuries, and compartment syndrome Intraneural ganglion cysts, tumors, such as lipoma, schwannoma, neurofibroma, osteochondroma, neurogenic sarcoma, focal hypertrophic neuropathy, desmoid tumor, or bone tumors, hematoma, and abscess Weight loss in patients with prolonged bed rest Deep venous thrombosis
Treatment of underlying cause, physiotherapy, analgesic agents, neurolysis, or nerve grafting Treatment for mass lesions, decompression of the nerve, and analgesic agents
Physiotherapy and analgesic agents
Treatment for venous thrombosis, elevation, analgesic agents, and anticoagulation Knee deformity and associated nerve stretch Orthopedic management, physiotherapy, and analgesic agents True or false arterial aneurysms or limb Decompression of the mass lesion, vascular injury repair, physiotherapy, and analgesic agents Antineoplastic agents Change of antineoplastic agent Isolated nerve palsy Treatment of systemic disorder and analgesic agents
evaluation. Briefly, a single radiograph and computed tomography scan can help to establish anatomic relationships between exostosis, pseudoaneurysm, and the affected artery. The diagnosis of pseudoaneurysm is generally made by color Duplex ultrasonography. Nevertheless, contrast-enhanced computed tomography and magnetic resonance imaging studies are helpful to define the relationship between the exostosis and pseudoaneurysm of the popliteal artery. A 3-dimensional reconstruction of the images also gives spatial information of the lesions. The management of nerve compression syndromes depends on the etiology11e19 (Table I). The removal of the predisposing cause, lesion, or activity is the best treatment of peroneal neuropathies.6 In pseudoaneurysm of the popliteal artery, it has been noted that the initial treatment of peroneal nerve palsy requires immediate repair of the aneurysm and decompression of the nerve.3e5 If there is no clinical improvement, surgical intervention for neuropathy is then recommended. Neurolysis, nerve grafting, or a combination of the two is considered if there is partial or complete nerve injury, especially in cases of trauma. Garazzo et al.20 reported that common peroneal nerve palsies in open wounds should undergo surgical exploration at emergency. In closed injuries with no spontaneous recovery within 4 months postinjury, patients should be referred for surgical treatment regardless of the causative mechanism of the lesion. In addition, physical
therapy is mandatory to prevent contracture or to relieve deformities caused by a loss of or weakness in the range of motion of the joints. Clinically, pain is considered to be the early symptom in peroneal neuropathy. Its management may be difficult with conventional analgesic agents, such as acetaminophen or nonsteroidal antiinflammatory drugs. Alternative analgesic agents to relieve neuropathic pain, such as antiepileptic drugs, opioids, or selective serotonin uptake inhibitors can be necessary.6 In our patient, gabapentin provided effective postoperative analgesia. Surgical treatment of the vascular complications of exostosis is recommended as an urgent procedure to avoid arterial occlusion, embolism, or phlebitis.10 Pseudoaneurysms can be repaired with primary closure of defect, reconstruction of the artery with a vein patch, resection of the artery and end to end anastomosis, venous grafting, or polytetrafluoroethylene bypass. The pseudoaneurysm sac, including blood and hematoma, is evacuated to decompress the peroneal nerve. With regard to surgical exposure, 3 approaches can be used for popliteal aneurysms: medial, posterior, and lateral. In our case, we used a medial approach to allow for simultaneous control of the common femoral, superficial femoral, and infrapopliteal arteries in the supine position before opening the pseudoaneurysm sac. This approach also enabled exploration of the bony spikes that were closely associated with
Vol. 28, No. 4, May 2014
the popliteal artery. The posterior approach could have been used in this patient, but flexion deformity of the knee joint might have limited adequate exposure of the sac and popliteal artery. In the published literature, there is no clear indication for the timing of excision of the exostosis. Surgical excision has been recommended in painful and enlarging lesions. Some authors recommend surgical repair of the exostosis if it becomes malignant; however, early excision is recommended if the exostosis is next to a major vessel or there is bone fracture or neurovascular injury.10 In our case, after repair of the popliteal artery with an end to end anastomosis, bony spikes were excised. The bony spikes were resected after reconstruction of the artery because there was an enough space in the popliteal fossa after evacuation of the pseudoaneurysm. Nevertheless, in some cases, an intraoperative arterial shunt may be considered to maintain temporary revascularization until the orthopedic portion of the case is completed. In conclusion, peroneal nerve palsy is an unusual complication of popliteal pseudoaneurysms. Patients with hereditary multiple exostoses should be followed-up closely to prevent associated vascular and neurologic injuries. REFERENCES 1. Dawson DM, Hallett M, Wilbourn AJ. Entrapment neuropathies. 3rd ed. Philadelphia: Lippincott-Raven, 1999. 2. McCluskey LF, Webb LB. Compression and entrapment neuropathies of the lower extremity. Clin Podiatr Med Surg 1999;16:97e125. 3. Ersozlu S, Ozulku M, Yildirim E, et al. Common peroneal nerve palsy from an untreated popliteal pseudoaneurysm after penetrating injury. J Vasc Surg 2007;45:408e10. 4. Ozc¸akar L, Aknc A, Aksoy DY, et al. Peroneal neuropathy due to a popliteal aneurysm in a patient with infectious endocarditis. Ann Vasc Surg 2004;18:115e7.
Case reports 1037.e9
5. Megalopoulos A, Vasiliadis K, Siminas S, et al. Pseudoaneurysm of the popliteal artery complicated by peroneal mononeuropathy in a 4-year-old child: report of a case. Surg Today 2007;37:798e801. 6. Baima J, Krivickas L. Evaluation and treatment of peroneal neuropathy. Curr Rev Musculoskelet Med 2008;1:147e53. 7. Pavic P, Vergles D, Sarlija M, et al. Pseudoaneurysm of the popliteal artery in a patient with multiple hereditary exostoses. Ann Vasc Surg 2011;25:268.e1e2. 8. Lieberman J, Mazzucco J, Kwasnik E, et al. Popliteal pseudoaneurysm as a complication of an adjacent osteochondroma. Ann Vasc Surg 1994;8:198e203. 9. Matsushita M, Nishikimi N, Sakurai T, et al. Pseudoaneurysm of the popliteal artery caused by exostosis of the femur: case report and review of the literature. J Vasc Surg 2000;32:201e4. 10. Vasseur MA, Fabre O. Vascular complications of osteochondromas. J Vasc Surg 2000;31:532e8. 11. Kim DH, Murovic JA, Teil RL, et al. Management and outcomes in 318 operative common peroneal nerve lesions at the LSU Health Sciences Center. Neurosurgery 2004;54:1421e9. 12. Kim JY, Ihn YK, Kim JS, et al. Nontraumatic peroneal nerve palsy: MRI findings. Clin Radiol 2007;62:58e64. 13. Sotaniemi KA. Slimmer’s paralysisdperoneal neuropathy during weight reduction. J Neurol Neurosurg Psychiatry 1984;47:564e6. 14. Katirji MB, Wilbourn AJ. Common peroneal mononeuropathy: a clinical and electrophysiologic study of 116 lesions. Neurology 1988;38:1723e8. 15. Fukuda H. Bilateral peroneal nerve palsy caused by intermittent pneumatic compression. Intern Med 2006;45:93e4. 16. Fetzer B, Prather H, Gelberman RH, et al. Progressive peroneal nerve palsy in a varus arthritic knee. A case report. J Bone Joint Surg Am 2004;86:1538e40. 17. Argyriu AA, Bruna J, Marmiroli P, et al. Chemotherapyinduced peripheral neurotoxicity (CIPN): an update. Crit Rev Oncol Hematol 2012;82:51e77. 18. Wolf J, Schmitt V, Palm F, et al. Peripheral neuropathy as initial manifestation of primary systemic vasculitides. J Neurol 2013;260:1061e70. 19. Florica B, Aghdassi E, Su J, et al. Peripheral neuropathy in patients with systemic lupus erythematosus. Semin Arthritis Rheum 2011;41:203e11. 20. Garozzo D, Ferraresi S, Buffatti P. Surgical treatment of common peroneal nerve injuries: indications and results. A series of 62 cases. J Neurosurg Sci 2004;48:105e12.