CLINICAL SPOTLIGHT
Heart, Lung and Circulation (2014) 23, e255–e257 1443-9506/04/$36.00 http://dx.doi.org/10.1016/j.hlc.2014.07.067
A Case of Popliteal Artery Entrapment Syndrome Presenting with Acute Limb Ischaemia Byeong-joo Jo, MD, Jun-Ho Bae, MD, PhD * Division of Cardiology, Department of Internal Medicine, College of Medicine, Dongguk University, Gyeongju, South Korea Received 5 June 2014; accepted 21 July 2014; online published-ahead-of-print 4 August 2014
Popliteal artery entrapment syndrome (PAES) is caused by an anomalous anatomic relationship between the popliteal artery and the musculotendinous structures in the popliteal fossa. In the early stage of PAES, patients most commonly present with intermittent claudication. If we don’t perform appropriate investigation, the diagnosis of PAES is easily missed and may lead to irreversible arterial damage. Thrombus formation in the damaged popliteal artery can cause complete obstruction of the popliteal artery leading to acute limb-threatening ischaemia. Keywords
Popliteal artery Popliteal artery entrapment syndrome Acute limb ischaemia Intermittent claudication Peripheral vascular disease
Introduction
Case report
PAES is a rare but potentially limb threatening peripheral vascular disease occurring predominantly in young adults [1]. Popliteal artery and vein are normally located between the two heads of the gastrocnemius muscle. Abnormalities in this relationship can produce PAES [2]. Due to the complexity of the embryologic development, PAES has been classified as various types according to the anatomical abnormalities [3–5]. These abnormal anatomic relationships can produce extrinsic compression of the popliteal artery and cause vascular damage [6]. The PAES is characterised by calf pain and intermittent claudication in persons without atherosclerosis, mostly young men [7]. If patients have not developed sufficient collateral circulation in the later stages of PAES, thrombus formation may cause complete obstruction of the popliteal artery leading to acute limb-threatening ischaemia. This syndrome is hard to diagnose, therefore careful history and physical examination is required combined with imaging testing. We report a case of PAES presenting with acute limb ischaemia.
A 30 year-old man was admitted to the hospital with his right calf in pain. He was diagnosed with hypertension and was taking medications. He suffered from cramping pain of his right calf after strenuous exercise for about seven years. But, recently he complained about worsening pain of his right calf even following a rest. Not only pain but also claudication was his chief complaint. He was a smoker, and had hypertension and dyslipidaemia. Physical examination revealed a muscular young man, 86 kg in weight, 183 cm in height, with pulse rate 66/min and a blood pressure reading of 130/80 mmHg. Neither right dorsalis pedis artery nor right popliteal artery’s pulse was palpable. There was pale skin colour change; he could feel cold sensation on right lower leg. Ankle brachial index showed right 0.48 and left 1.08. On lower extremity computerised tomography angiography (CTA), medial displacement of the popliteal artery was noted and gastrocnemius medial head with lateral insertion on the distal femur was also identified (Fig. 1). CT scans at the level of popliteal fossa showed popliteal artery stenosis, post-stenotic
*Corresponding author at: Division of Cardiology, Department of Internal Medicine, Gyeongju Hospital, College of Medicine, Dongguk University, 87, Dongdaero, Gyeongju, Gyeongsangbuk-do, South Korea. Tel.: +82-54-770-8587; Fax: +82-54-770-8529., Email: [email protected] © 2014 Published by Elsevier Inc on behalf of Australian and New Zealand Society of Cardiac and Thoracic Surgeons (ANZSCTS) and the Cardiac Society of Australia and New Zealand (CSANZ).
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Figure 1 Peripheral CT angiography showed medial dislocation of popliteal artery and post stenotic dilatation to true aneurysm formation in anterior view (A). The medial head of the right gastrocnemius muscle (white arrow) arises from an abnormal lateral position in posterior view of 3D reconstruction CT image (B).
dilatation and aneurysm formation (Fig. 2). On axial view, right popliteal artery descended medial to right medial head of gastrocnemius muscle (Fig. 2). The medial head of the right gastrocnemius muscle arose from an abnormal lateral position in posterior view of 3D reconstruction CT image (Fig. 1). CT scans also showed thrombotic occlusion of right anterior, posterior tibial artery and peroneal artery. Blood test showed no abnormal findings. Following the diagnosis of PAES type2 presenting with acute limb ischaemia, which is characterised by abnormal gastrocnemius medial head with lateral insertion on the distal femur and medial displacement of the popliteal artery, he underwent surgery. During the surgical intervention, the medial head of the gastrocnemius muscle was resected, thrombus in popliteal artery was removed. Postoperatively, he had an uneventful postoperative course and was discharged on the fifth postoperative day. He started taking warfarin. At follow-up examinations after
discharge, the patient no longer complained of initial calf pain on his right leg.
Discussion Intermittent claudication is considered to be the classic manifestation of chronic arterial obstruction in the legs. In older patients suffering from atherosclerotic peripheral vascular disease, intermittent claudication is a common symptom. However, intermittent claudication of a lower extremity in a young patient is an unusual symptom and should prompt a search for causes other than those typically seen in older patients. The differential diagnosis include extrinsic compression by an osteochondroma or other bone lesion, PAES, cystic adventitial disease, fibromuscular dysplasia, Takayasu’s arteritis, peripheral arterial occlusive disease and Buerger’s disease [8]. PAES is an uncommon cause of lower-extremity
Figure 2 Axial view showed right popliteal artery (white arrow) descends medial to right medial head of gastrocenmius muscle (white open line).
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A Case of Popliteal Artery Entrapment Syndrome
claudication which usually occurs in younger patients who lack the risk factors for atherosclerosis and who are healthier and more active than average for their age group. The most widely accepted classification was made by Whelan and modified by Rich. According to this classification, PAES is classified into six types [3]. Type 2 is the abnormal gastrocnemius medial head with lateral insertion on the distal femur and medial displacement of the popliteal artery. PAES is a potentially limb threatening peripheral vascular disease [1]. If patients have not developed sufficient collateral circulation in the later stages of PAES, thrombus formation may cause complete obstruction of the popliteal artery leading to acute limb-threatening ischaemia. This syndrome is hard to diagnose, therefore careful history and physical examination is required combined with imaging testing. The imaging evaluation should begin with a radiograph of the affected extremity to exclude a bone lesion causing extrinsic compression of the nearby artery. The evaluation should also include an angiographic study to localise and characterise the arterial compromise. Noninvasive imaging techniques such as Doppler ultrasound, CT, CTA, magnetic resonance imaging (MRI) and magnetic resonance angiography (MRA) may be used for diagnosis. CT with or without 3D reconstruction is good for diagnosing PAES. MRI may be performed at the same time as MRA and provides optimal soft-tissue characterisation [9]. Surgery is a treatment option. Complete division of the abnormal muscle and transposition of the popliteal artery laterally may be all that is required in an undamaged artery. If the entrapped artery is damaged or occluded, then a bypass procedure using autologous vein is the procedure of choice. Catheter thromboembolectomy with intra-arterial thrombolysis has been reported as a viable option before surgical transposition if the situation permits [10,11]. In this case, the patient was diagnosed with type 2 PAES presenting with acute limb ischaemia by CT
angiography and the careful physical examination. And the patient is now free of lower extremity claudication with the help of surgery. In conclusion, the physician should consider PAES when the patients complain of intermittent claudication, especially when they are young. Early investigation and management can not only relieve the patient’s pain, but also save the limb from the risk of amputation.
References [1] Stavros Gourgiotis, John Aggelakas, Nikolaos Salemis, Charalabos Elias, Charalabos Georgiou. Diagnosis and surgical approach of popliteal artery entrapment syndrome: a retrospective study. Vasc Health Risk Manag 2008;4(1):83–8. [2] Wright Lonnie B, Matchett W Jean, Cruz Carlos P, James Charles A, Culp William C, Eidt John F, et al. McCowan. Popliteal Artery Disease: Diagnosis and Treatment. RadioGraphics 2004;24:467–79. [3] Rich NM, Collins Jr GJ, McDonal PT, Kozloff L, Clagett GP, Collins JT. Popliteal vascular entrapment: its increasing interest. Arch Surg 1979;114: 1377–84. [4] Stuart TP. Note on a variation in the course of the popliteal artery. J Anat Physiol 1879;13:162. [5] Johnsen JB, Holter O. Popliteal artery entrapment syndrome. Acta Chir Scand 1984;150:493–6. [6] Ikeda M, Iwase T, Ashida K, Tankawa H. Popliteal artery entrapment syndrome. Report of a case and study of 18 cases in Japan. Am J Surg 1981;141:726–30. [7] Zhong H, Gan J, Zhao Y, Xu Z, Liu C, Shao G, et al. Role of CT angiography in the diagnosis and treatment of popliteal vascular entrapment syndrome. Am J Roentgenol 2011 Dec;197(6):W1147–54. [8] Teresa L, Carman, Bernardo B, Fernandez JR. A Primary Care Approach to the Patient with Claudication. Am Fam Physician 2000;61(4):1027–32. [9] Sakamoto A, Tanaka K, Matsuda S, Harmaya K, Iwamoto Y. Vascular compression caused by solitary osteochondroma: useful diagnostic methods of magnetic resonance angiography and Doppler ultrasonography. J Orthop Sci 2002;7:439–43. [10] Steurer J, Hoffmann U, Schneider E, Largiader J, Bollinger A. A new therapeutic approach to popliteal artery entrapment syndrome. Eur J Vasc Endovasc Surg 1995;10:243–7. [11] Chan CWM, Wilson JI, Myatt A, Roberts PN. Painful leg and missing pulses: a case report. Arch Dis Child 2000;83:362–3.
Heart, Lung and Circulation (2014) 23, e255–e257 1443-9506/04/$36.00 http://dx.doi.org/10.1016/j.hlc.2014.07.067
A Case of Popliteal Artery Entrapment Syndrome Presenting with Acute Limb Ischaemia Byeong-joo Jo, MD, Jun-Ho Bae, MD, PhD * Division of Cardiology, Department of Internal Medicine, College of Medicine, Dongguk University, Gyeongju, South Korea Received 5 June 2014; accepted 21 July 2014; online published-ahead-of-print 4 August 2014
Popliteal artery entrapment syndrome (PAES) is caused by an anomalous anatomic relationship between the popliteal artery and the musculotendinous structures in the popliteal fossa. In the early stage of PAES, patients most commonly present with intermittent claudication. If we don’t perform appropriate investigation, the diagnosis of PAES is easily missed and may lead to irreversible arterial damage. Thrombus formation in the damaged popliteal artery can cause complete obstruction of the popliteal artery leading to acute limb-threatening ischaemia. Keywords
Popliteal artery Popliteal artery entrapment syndrome Acute limb ischaemia Intermittent claudication Peripheral vascular disease
Introduction
Case report
PAES is a rare but potentially limb threatening peripheral vascular disease occurring predominantly in young adults [1]. Popliteal artery and vein are normally located between the two heads of the gastrocnemius muscle. Abnormalities in this relationship can produce PAES [2]. Due to the complexity of the embryologic development, PAES has been classified as various types according to the anatomical abnormalities [3–5]. These abnormal anatomic relationships can produce extrinsic compression of the popliteal artery and cause vascular damage [6]. The PAES is characterised by calf pain and intermittent claudication in persons without atherosclerosis, mostly young men [7]. If patients have not developed sufficient collateral circulation in the later stages of PAES, thrombus formation may cause complete obstruction of the popliteal artery leading to acute limb-threatening ischaemia. This syndrome is hard to diagnose, therefore careful history and physical examination is required combined with imaging testing. We report a case of PAES presenting with acute limb ischaemia.
A 30 year-old man was admitted to the hospital with his right calf in pain. He was diagnosed with hypertension and was taking medications. He suffered from cramping pain of his right calf after strenuous exercise for about seven years. But, recently he complained about worsening pain of his right calf even following a rest. Not only pain but also claudication was his chief complaint. He was a smoker, and had hypertension and dyslipidaemia. Physical examination revealed a muscular young man, 86 kg in weight, 183 cm in height, with pulse rate 66/min and a blood pressure reading of 130/80 mmHg. Neither right dorsalis pedis artery nor right popliteal artery’s pulse was palpable. There was pale skin colour change; he could feel cold sensation on right lower leg. Ankle brachial index showed right 0.48 and left 1.08. On lower extremity computerised tomography angiography (CTA), medial displacement of the popliteal artery was noted and gastrocnemius medial head with lateral insertion on the distal femur was also identified (Fig. 1). CT scans at the level of popliteal fossa showed popliteal artery stenosis, post-stenotic
*Corresponding author at: Division of Cardiology, Department of Internal Medicine, Gyeongju Hospital, College of Medicine, Dongguk University, 87, Dongdaero, Gyeongju, Gyeongsangbuk-do, South Korea. Tel.: +82-54-770-8587; Fax: +82-54-770-8529., Email: [email protected] © 2014 Published by Elsevier Inc on behalf of Australian and New Zealand Society of Cardiac and Thoracic Surgeons (ANZSCTS) and the Cardiac Society of Australia and New Zealand (CSANZ).
e256
B.-j. Jo, J.-H. Bae
Figure 1 Peripheral CT angiography showed medial dislocation of popliteal artery and post stenotic dilatation to true aneurysm formation in anterior view (A). The medial head of the right gastrocnemius muscle (white arrow) arises from an abnormal lateral position in posterior view of 3D reconstruction CT image (B).
dilatation and aneurysm formation (Fig. 2). On axial view, right popliteal artery descended medial to right medial head of gastrocnemius muscle (Fig. 2). The medial head of the right gastrocnemius muscle arose from an abnormal lateral position in posterior view of 3D reconstruction CT image (Fig. 1). CT scans also showed thrombotic occlusion of right anterior, posterior tibial artery and peroneal artery. Blood test showed no abnormal findings. Following the diagnosis of PAES type2 presenting with acute limb ischaemia, which is characterised by abnormal gastrocnemius medial head with lateral insertion on the distal femur and medial displacement of the popliteal artery, he underwent surgery. During the surgical intervention, the medial head of the gastrocnemius muscle was resected, thrombus in popliteal artery was removed. Postoperatively, he had an uneventful postoperative course and was discharged on the fifth postoperative day. He started taking warfarin. At follow-up examinations after
discharge, the patient no longer complained of initial calf pain on his right leg.
Discussion Intermittent claudication is considered to be the classic manifestation of chronic arterial obstruction in the legs. In older patients suffering from atherosclerotic peripheral vascular disease, intermittent claudication is a common symptom. However, intermittent claudication of a lower extremity in a young patient is an unusual symptom and should prompt a search for causes other than those typically seen in older patients. The differential diagnosis include extrinsic compression by an osteochondroma or other bone lesion, PAES, cystic adventitial disease, fibromuscular dysplasia, Takayasu’s arteritis, peripheral arterial occlusive disease and Buerger’s disease [8]. PAES is an uncommon cause of lower-extremity
Figure 2 Axial view showed right popliteal artery (white arrow) descends medial to right medial head of gastrocenmius muscle (white open line).
e257
A Case of Popliteal Artery Entrapment Syndrome
claudication which usually occurs in younger patients who lack the risk factors for atherosclerosis and who are healthier and more active than average for their age group. The most widely accepted classification was made by Whelan and modified by Rich. According to this classification, PAES is classified into six types [3]. Type 2 is the abnormal gastrocnemius medial head with lateral insertion on the distal femur and medial displacement of the popliteal artery. PAES is a potentially limb threatening peripheral vascular disease [1]. If patients have not developed sufficient collateral circulation in the later stages of PAES, thrombus formation may cause complete obstruction of the popliteal artery leading to acute limb-threatening ischaemia. This syndrome is hard to diagnose, therefore careful history and physical examination is required combined with imaging testing. The imaging evaluation should begin with a radiograph of the affected extremity to exclude a bone lesion causing extrinsic compression of the nearby artery. The evaluation should also include an angiographic study to localise and characterise the arterial compromise. Noninvasive imaging techniques such as Doppler ultrasound, CT, CTA, magnetic resonance imaging (MRI) and magnetic resonance angiography (MRA) may be used for diagnosis. CT with or without 3D reconstruction is good for diagnosing PAES. MRI may be performed at the same time as MRA and provides optimal soft-tissue characterisation [9]. Surgery is a treatment option. Complete division of the abnormal muscle and transposition of the popliteal artery laterally may be all that is required in an undamaged artery. If the entrapped artery is damaged or occluded, then a bypass procedure using autologous vein is the procedure of choice. Catheter thromboembolectomy with intra-arterial thrombolysis has been reported as a viable option before surgical transposition if the situation permits [10,11]. In this case, the patient was diagnosed with type 2 PAES presenting with acute limb ischaemia by CT
angiography and the careful physical examination. And the patient is now free of lower extremity claudication with the help of surgery. In conclusion, the physician should consider PAES when the patients complain of intermittent claudication, especially when they are young. Early investigation and management can not only relieve the patient’s pain, but also save the limb from the risk of amputation.
References [1] Stavros Gourgiotis, John Aggelakas, Nikolaos Salemis, Charalabos Elias, Charalabos Georgiou. Diagnosis and surgical approach of popliteal artery entrapment syndrome: a retrospective study. Vasc Health Risk Manag 2008;4(1):83–8. [2] Wright Lonnie B, Matchett W Jean, Cruz Carlos P, James Charles A, Culp William C, Eidt John F, et al. McCowan. Popliteal Artery Disease: Diagnosis and Treatment. RadioGraphics 2004;24:467–79. [3] Rich NM, Collins Jr GJ, McDonal PT, Kozloff L, Clagett GP, Collins JT. Popliteal vascular entrapment: its increasing interest. Arch Surg 1979;114: 1377–84. [4] Stuart TP. Note on a variation in the course of the popliteal artery. J Anat Physiol 1879;13:162. [5] Johnsen JB, Holter O. Popliteal artery entrapment syndrome. Acta Chir Scand 1984;150:493–6. [6] Ikeda M, Iwase T, Ashida K, Tankawa H. Popliteal artery entrapment syndrome. Report of a case and study of 18 cases in Japan. Am J Surg 1981;141:726–30. [7] Zhong H, Gan J, Zhao Y, Xu Z, Liu C, Shao G, et al. Role of CT angiography in the diagnosis and treatment of popliteal vascular entrapment syndrome. Am J Roentgenol 2011 Dec;197(6):W1147–54. [8] Teresa L, Carman, Bernardo B, Fernandez JR. A Primary Care Approach to the Patient with Claudication. Am Fam Physician 2000;61(4):1027–32. [9] Sakamoto A, Tanaka K, Matsuda S, Harmaya K, Iwamoto Y. Vascular compression caused by solitary osteochondroma: useful diagnostic methods of magnetic resonance angiography and Doppler ultrasonography. J Orthop Sci 2002;7:439–43. [10] Steurer J, Hoffmann U, Schneider E, Largiader J, Bollinger A. A new therapeutic approach to popliteal artery entrapment syndrome. Eur J Vasc Endovasc Surg 1995;10:243–7. [11] Chan CWM, Wilson JI, Myatt A, Roberts PN. Painful leg and missing pulses: a case report. Arch Dis Child 2000;83:362–3.
