Popliteal Artery Cystic Adventitial Disease: Early Lessons in Treatment Shantanu Warhadpande, Michael R. Go, Hosam El Sayed, Bhagwan Satiani, and Patrick S. Vaccaro, Columbus, Ohio
Background: We present 6 patients who had operative repair of symptomatic popliteal cystic adventitial disease (pCAD). Developmental theories for pCAD and surgical alternatives are presented. Methods: All patients who had repair of pCAD over the past 3 years are included. Results: Three patients had cyst excision alone, whereas the remaining 3 had cyst and artery excision with interposition vein grafting. Cyst recurrence occurred in 2 patients who had cyst excision alone. Four of the patients had a patent communication between the cyst and the joint capsule. Conclusions: Our small series suggests that the articular (synovial) theory of development may be the most likely and that cyst and artery excision with interposition vein grafting may be preferred over cyst excision alone.
Cystic adventitial disease (CAD) is rare and characterized by mucinous cyst formation within the arterial or venous adventitial layer. The popliteal artery is the most commonly affected artery in CAD, and much of the existing literature focuses on the pathogenesis, management, and treatment of popliteal artery CAD (pCAD). This mucinous cyst can compress the popliteal artery and lead to a focal luminal stenosis causing intermittent claudication. The best treatment remains controversial, but the choice is usually either cyst excision alone or cyst and artery excision with interposition grafting. We present 6 patients with pCAD, half of whom were treated with cyst excision and half with cyst and popliteal artery resection with saphenous vein reconstruction. This small experience suggests that cyst and artery excision with interposition grafting may be the better option.
Division of Vascular Diseases and Surgery, The Ohio State University Wexner Medical Center, Columbus, OH. Correspondence to: Patrick S. Vaccaro, MD, MBA, 701 Prior Hall, 376 West 10th Avenue, Columbus, OH 43210, USA; E-mail: patrick.
[email protected] Ann Vasc Surg 2017; 38: 255–259 http://dx.doi.org/10.1016/j.avsg.2016.05.112 Ó 2016 Elsevier Inc. All rights reserved. Manuscript received: February 10, 2016; manuscript accepted: May 10, 2016; published online: 12 August 2016
METHODS Six patients were identified who had repair of pCAD at The Ohio State University during the past 3 years. Observations were drawn from these 6 clinical presentations. Patient 1 This 48-year-old man, an avid runner, presented to the vascular surgery clinic with a recent 3-week history of right foot claudication and associated numbness and tingling in the toes. His physical examination was significant for a cold right calf and foot. He had a weakly palpable posterior tibial pulse on the right and no dorsalis pedis pulse. Left pedal pulses were normal. A subsequent arterial duplex showed a 1.0 2.48-cm hypoechoic collection in the right popliteal fossa along with a stenotic area in the popliteal artery (Fig. 1). The tibial arteries were all patent. His ankle-brachial index (ABI) at the right posterior tibial artery was 1.13 but reduced to 0.99 at the dorsalis pedis artery. ABIs on the left were normal. He underwent an uncomplicated cyst excision through a posterior approach. The cyst was singular and discrete. A posterolateral connection from the cyst to the joint space was identified and ligated flush with the joint capsule (Fig. 2). An intraoperative completion angiogram 255
256 Warhadpande et al.
Annals of Vascular Surgery
Fig. 1. Duplex scan of right popliteal artery of patient 1 with bold arrow pointing to cyst and narrow artery showing the compressed popliteal artery.
showed brisk flow through the popliteal artery with no stenotic areas. The patient has been followed for 36 months and has had no recurrence of symptoms with ABIs of 1.16 and triphasic pedal waveforms. Patient 2 A healthy 34-year-old woman presented to the vascular clinic with a 1-year history of left calf claudication. Pulse examination and ABIs were normal. A subsequent magnetic resonance imaging (MRI) demonstrated a multilobulated popliteal artery cyst causing extrinsic compression (>50%) of the artery and a small Baker cyst (Fig. 3). The initial report described this as a ganglion cyst causing the extrinsic compression. Through a posterior approach, she underwent a very complicated popliteal artery cyst excision. Intraoperatively, there was significant cystic burden, and the cyst completely encased the popliteal artery circumferentially. A connection between the joint capsule and the cyst was identified and ligated (Fig. 4). The cyst wall was then resected off the popliteal artery along the length and circumference of the artery. The popliteal artery was ensured to be of normal caliber and with a strong pulse. Several smaller cysts attached to the gastrocnemius tendon were not excised because they did not involve the artery. Her postoperative course was uncomplicated. Six months after her operation, however, the patient
Fig. 2. Arrow highlighting connection between cyst and joint capsule in patient 1.
returned with fullness in her left popliteal fossa but without recurrent claudication. MRI showed multiple cystic lesions in her popliteal fossa but no arterial compression (Fig. 5). She persisted in complaining of a sense of fullness and pressure in the popliteal fossa from the recurrent cysts although the popliteal artery was not involved. She was referred to orthopedic surgery for further evaluation, and they excised a torn medial meniscus but left the cysts alone. At 26 months, she remains free of claudication but continues to complain of fullness and pressure.
Volume 38, January 2017
Popliteal artery cystic adventitial disease 257
Fig. 3. MRI of knee in patient 2 (arrow) showing multilobulated cyst compressing the popliteal artery.
Fig. 5. Postoperative MRI of patient 2 showing recurrent cysts (arrow) but no further compression on the popliteal artery.
of pain with ambulation relieved by rest. CTA visualized a cystic structure overlying both the popliteal artery and vein throughout the length of the previous excision (Fig. 6). She refused further intervention and has not returned for follow-up. Patient 4
Fig. 4. View through cyst (arrow) at orifice of communication between cyst and joint capsule in patient 2.
Patient 3 A 50-year-old woman presented with claudication, and a computed tomography angiography (CTA) showed cystic compression of the popliteal artery from above the knee to the trifurcation. There were multiple cystic structures compressing the arterial lumen. She recently had an unsuccessful attempt at percutaneous cyst aspiration and was referred for cyst excision. The popliteal artery adventitial cyst was excised from the above-knee segment as far distally as possible. The tibial nerve made further excision dangerous, and the small remaining cyst capsule was opened but not completely excised. No connection from the cyst to the joint space was identified. The patient returned to the vascular surgery clinic 4 months later completely free of claudication. However, at 10 months, she returned complaining of new-onset claudication. As before, she complained
A 53-year-old man was seen in consultation for calf claudication after walking 100 yards. His physical examination was noncontributory. An MRI confirmed the presence of a large cystic mass overlying the popliteal artery. Intraoperatively, a communication between the cystic mass and the joint space was identified and ligated. The entire cystic mass, along with the underlying popliteal artery, was removed and replaced with great saphenous vein. The patient had strong distal pulses postoperatively and at 8month follow-up had no recurrence of symptoms. Patient 5 A 45-year-old man had very short distance claudication and numbness of the toes of the left foot as the claudication worsened. Angiography showed a ‘‘scimitar’’ sign in the proximal left popliteal artery (Fig. 7). A direct communication from the cyst to the joint capsule was ligated and divided. The entire cyst and underlying popliteal artery were excised and replaced with saphenous vein. The patient remained free of claudication with normal ABIs at his 6-month postoperative visit.
258 Warhadpande et al.
Annals of Vascular Surgery
RESULTS AND DISCUSSION
Fig. 6. Arrow pointing to recurrent cyst on CTA in patient 3.
Fig. 7. Contrast angiogram showing compression of popliteal artery (arrow) by pCAD in patient 5.
Patient 6 A 31-year-old woman presented with calf claudication at short distances. One year before, she underwent balloon angioplasty of the popliteal artery for a high-grade stenosis. She noted improvement in her claudication for 8 months, but then symptoms recurred. An MRI showed narrowing of the popliteal artery by a cystic mass, suggestive of adventitial cystic disease. She had excision of the cyst and artery with a saphenous interposition graft. There was no communication noted between the cyst and joint capsule. At 5 months, she is free of claudication with normal pedal pulses and ABIs.
We present 6 cases of pCAD. The pathophysiology of CAD has not been fully delineated, and there are 4 different theories. The traumatic theory suggests that repeated trauma to the artery results in cystic adventitial degeneration.1,2 The systemic process theory asserts that cystic adventitial degeneration occurs as a result of a systemic process.1,2 The developmental theory states that mesenchymal cells from neighboring joints are incorporated within the adventitia during development; these mesenchymal cells then differentiate into mucin-producing cells.3e6 The final theory, the articular (synovial) theory, has recently gained significant traction. Presented by Spinner et al.,7 the articular theory contends that the cyst originates within the joint space and dissects within articular branches to the main artery. This articular connection between the adventitial cyst and the joint space allows propagation of mucinous fluid into the cyst, which histologically is not distinguishable from a ganglion cyst.5 Desy and Spinner2 further corroborated the articular theory in their review of 724 cases. Four of our six patients had a communication between the cyst and the joint capsule, lending support to the articular theory. This communication is easy to identify if dissection remains in contact with the cyst capsule. The connections is a tubular structure composed of tough synovial tissue that is easily followed from the cyst to the joint capsule, and the patency is confirmed by the passage of a small metallic probe or Fogarty catheter from the open cyst to the joint capsule (Fig. 2). Synovial fluid is expressible from the joint capsule by the application of light pressure. Our numbers are not large enough to determine if lack of identification and ligation of the connection is an independent risk factor for recurrence. Three of our patients were treated with cyst excision alone and 3 with cyst plus popliteal artery resection and saphenous vein interposition grafting. Of the 3 patients treated with cyst excision alone, 2 had recurrence of their pCAD. Furthermore, in one of the recurrences after cyst excision (patient 2), a connection between the joint space and cyst was identified and ligated. However, the cyst recurrence did not involve the artery but did involve the surrounding muscle tendon. The other patient who had a recurrence after cyst excision alone (patient 3) did not have her joint connection ligated. Patients 4, 5, and 6 who had cyst plus artery resection have remained recurrence free and asymptomatic. In addition to the lack of consensus on the pathogenesis of CAD, management and treatment of CAD vary. Treatment options include cyst excision alone,
Volume 38, January 2017
cyst and underlying vessel resection with vessel reconstruction using vein autograft, cyst aspiration, and angioplasty.2 The review article by Desy and Spinner studied the efficacy of various treatment options and found that percutaneous treatment of CAD (cyst aspiration and angioplasty) was a significant risk factor in cyst recurrence (OR, 13.7). Of the cases that received angioplasty and percutaneous aspiration for CAD, 67% and 47% had cyst recurrence, respectively.2 Cyst excision alone resulted in a recurrence rate of 7% (10 arterial cyst recurrences of 151 cyst excisions). Cyst resection with saphenous vein reconstruction resulted in a recurrence rate of 1% (2 recurrences in 202 resections).2 By far, the most common treatment approach was cyst and artery resection with saphenous vein interposition, with nearly 40% of patients being treated as such.2 Comparatively, 22.8% of CAD patients were treated with cyst excision alone.2 Based on our experience with several cases, cyst excision alone may not be an appropriate surgical approach for patients with pCAD, given the recurrences we have seen in patients after this approach (Table I). The recurrences were at 6 and 10 months from the initial cyst excision. We have followed our patients at 1, 3, 6, and 12 months after operation with imaging done only for new or recurrent symptoms. ABIs were obtained at 6 and 12 months. Based on our earliest recurrence, duplex scanning of the popliteal artery should be considered at the 6-month visit along with the ABIs in all cases no matter the intervention, especially if symptoms recur. Even when, as per Desy’s articular theory of pathogenesis, a cyst-joint-space connection was ligated, 1 of the patients (patient 2) had a recurrence of her pCAD 6 months later. Thus, cyst plus artery resection with great saphenous vein bypass may be a better option and results in lower recurrence rates.2 This small series suggests that cyst excision alone may have a limited role in short-segment disease not involving the entire circumference. The comprehensive review by Desy and Spinner2 shows that excision with interposition grafting is the most frequent intervention with the lowest recurrence rate. We believe that this should be the operative choice in all situations unless it is not technically feasible.
Popliteal artery cystic adventitial disease 259
Table I. Cyst location by patient, intervention performed, development of recurrence and interval if present Patient Cyst location
1 2 3 4 5 6
Intervention
Recurrence/ time
Popliteal artery Cyst excision alone No Popliteal artery Cyst excision alone Yes/6 months Popliteal artery Cyst excision alone Yes/10 months Popliteal artery Excision and graft No Popliteal artery Excision and graft No Popliteal artery Excision and graft No
CONCLUSIONS In summary, our series suggests cyst and artery resection with an interposition vein graft may be a more thorough treatment than cyst excision alone. As seen in patients 2 and 3, cyst excision can result in early recurrence especially if the excision is incomplete. This study gives further support to the articular theory of cyst development because twothirds of our patients had a communication between the cyst and the joint. REFERENCES 1. Ishikawa K. Cystic adventitial disease of the popliteal artery and of other stem vessels in the extremities. Jpn J Surg 1987;17:221e9. 2. Desy NM, Spinner RJ. The etiology and management of cystic adventitial disease. J Vasc Surg 2014;60:235e45. 245.e1e11. 3. Tsolakis IA, Walvatne CS, Caldwell MD. Cystic adventitial disease of the popliteal artery: diagnosis and treatment. Eur J Vasc Endovasc Surg 1998;15:188e94. 4. Hernandez Mateo MM, Serrano Hernando FJ, L opez IM, et al. Cystic adventitial degeneration of the popliteal artery: report on three cases and review of the literature. Ann Vasc Surg 2014;28:1062e9. 5. Levien LJ, Benn CA. Adventitial cystic disease: a unifying hypothesis. J Vasc Surg 1998;28:193e205. 6. Flanigan DP, Burgham SJ, Goodreau JJ, Bergan JJ. Summary of cases of adventitial cystic disease of the popliteal artery. Ann Surg 1979;189:165e75. 7. Spinner RJ, Desy NM, Agarwal G, et al. Evidence to support that adventitial cysts, analogous to intraneural ganglion cysts, are also joint-connected. Clin Anat 2013;26:267e81.