Ischemic Stroke due to Paradoxical Embolism Arising from a Popliteal Vein Aneurysm Laurent Auboire,1 Laura Palcau,2 Elise Mackowiak,2 Fausto Viader,3,4 Claire Le Hello,1 and Ludovic Berger,2,4 Caen, France
Popliteal vein aneurysm (PVA) is a very uncommon cause of stroke. We present the case of a 63-year-old, right-handed man who presented to the emergency department with transitory ischemic accident (left superior extremity regressive monoplegia). Complete etiologic work-up led to the discovery of both a patent foramen ovale associated with an septum interauricular aneurysm, and of a PVA without mural thrombus. The diagnosis of brain paradoxical embolism was retained. The unique potentially embolic cause was the presence of the PVA. We decided to treat the PVA surgically to avoid a further cerebral vascular episode. Although uncommon, venous etiology must be considered for stroke.
Certain strokes of unknown etiology may be the result of a paradoxical embolism traversing through a patent foramen ovale (PFO).1 Some 25% of the healthy adult population is known to have a PFO, rarely accompanied by a septum interauricular aneurysm (SIA).2 Observational studies have established a strong association between the presence of PFO with SIA and increased risk of ischemic stroke.2,3 Some of these may be caused by paradoxical embolism from a venous thrombus that travels via the PFO to the systemic circulation,4 the source being a popliteal vein aneurysm (PVA).
Primary PVA is a rare condition (0.2%).5,6 Almost all patients reported in the literature are symptomatic, the most common symptom being pulmonary embolism (PE). May and Nissl7 were the first to describe a PVA in 1968. In 1976, Dahl et al.8 described thrombosis and PE as the most serious and common complication of PVA. A review of the literature conducted by Nasr et al.9 in 2008 included 120 cases of PVA and found that its most common presentation is through thromboembolic complications. Stroke due to paradoxical embolism in the presence of a PFO associated or not with SIA remains a rare entity (3.8e4.4%).2 We report a case of stroke due to a paradoxical embolism from PVA in a patient with PFO and SIA, a situation rarely reported.
1 Department of Vascular Medicine, Centre Hospitalier Universitaire de Caen, Caen, France. 2 Department of Vascular Surgery, Centre Hospitalier Universitaire de Caen, Caen, France.
CASE REPORT
3 Department of Neurology, Centre Hospitalier Universitaire de Caen, Caen, France.
A 63-year-old, right-handed man presented to the emergency department for a transient ischemic accident. He complained of transitory numbness of the left superior limb. His medical history included left central retinal artery occlusion. Cardiovascular risk factors were dyslipidemia and smoking. At the time of presentation he was on clopidogrel for a history of retinal artery occlusion. Clinical examination was normal. We detected no carotid or heart murmur. Complete biologic tests were normal. Brain computed tomography (CT) scan revealed no abnormalities. Cerebral magnetic resonance imaging
4 University of Caen, Faculty of Medicine, Universite de Caen Bassee Normandie, Caen, France.
Correspondence to: Ludovic Berger, MD, Department of Vascular Surgery, Centre Hospitalier Universitaire de Caen, Avenue de la C^ote de Nacre, 14000 Caen, France; E-mail:
[email protected] Ann Vasc Surg 2014; 28: 738.e15–738.e17 http://dx.doi.org/10.1016/j.avsg.2013.06.021 Ó 2014 Elsevier Inc. All rights reserved. Manuscript received: May 5, 2013; manuscript accepted: June 19, 2013; published online: December 19, 2013.
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(MRI) showed a superficial right sylvian stroke. Supraaortic arterial trunk Doppler ultrasound and electrocardiogram were normal. Transesophageal echocardiogram showed an 8-mm PFO with bidirectional shunt and a 10-mm septum interauricular aneurysm. A venous Doppler ultrasound revealed a right 23-mm fusiform PVA with no mural thrombus. The patient underwent CT angiography, which confirmed the PVA (Fig. 1). We decided to perform surgical treatment of the PVA to avoid a further cerebral vascular episode. The surgical technique consisted of tangential aneurysmectomy with lateral venorrhaphy (Fig. 2). In this technique, the PVA is exposed through a standard posterior incision. Once exposed, the thin aneurysm sac could be easily differentiated from the normal vein wall and a proximal and distal complete clamping then realized. After sac incision, partially adherent mural thrombus was found in the dilated part of the vein and removed. The dilated part of the vein was excised and a double-layer suture was performed using 6-0 vascular suture materials. The postoperative course was uneventful. The patient received perioperative and 1-month postoperative anticoagulation (tinzaparin 4500 IU/day) once daily. No oral anticoagulation was initiated; only the antiplatelet therapy was continued. Immediately postoperatively the patient had a compressive stocking during the first month and ambulation was started at day 1. The patient had no further neurologic episode. At 6month follow-up, the clinical examination was normal, with a modified Rankin scale score of 0. Control Duplex ultrasound showed no abnormalities.
DISCUSSION PVA is an uncommon but potentially lifethreatening condition, as it can be a source of PE. Given the rarity of symptoms directly related to the aneurysm, this can be identified by physical examination or accompanied by leg pain and swelling in rare cases.10 In the presence of a PFO, inversion of the pressure in the atrial cavities (due to a cough or a Valsalva maneuver) may permit venous thrombus to pass through the left atrium and cause an ischemic stroke, known as a cold cerebral paradoxical embolism. The PFO is the most frequent abnormality associated with TIA in the adult patient. Four to 15% of patients with paradoxical embolism in the presence of a PFO will present with SIA. These abnormalities are easily diagnosed by transesophageal echocardiogram. Transcranial Doppler ultrasound with a microbubbles test is useful for detecting these abnormalities.2 Regarding PVA diagnosis, ascending venography was the choice imaging technique in earlier reports. Venous duplex scanning is currently considered the best noninvasive diagnostic method, because it gives
Annals of Vascular Surgery
Fig. 1. CT angiography showing the right retroarticular popliteal vein aneurysm.
both the real size of the aneurysm and the extent of any thrombus located within the aneurysmal sac. More recently, other diagnostic methods, such as CT scanning and MRI, have also been used and offer valuable anatomic information.11 Although large or saccular venous aneurysms are recognized to be more prone to thromboembolic complications,12 it seems that small aneurysms carry the same risk.13 Rupture of the PVA has never been reported in the literature. In our case, the duplex ultrasound did not identify any mural thrombus of the PVA, but complete etiologic testing revealed the only cause of the TIA was the presence of the PVA associated with PFO and SIA. Unlike Doppler ultrasound data, perioperative mural thrombus was found, confirming our diagnosis. In symptomatic patients with PE, or as in our case, with paradoxical embolism, surgery is the treatment of choice.14 Anticoagulation alone has proved to be inadequate in patients with PE and carries a high incidence (80%) of recurrence.10 Recurrence of PE after surgery has never been reported. Tangential aneurysmectomy with lateral venorrhaphy is the most commonly performed procedure. Management of asymptomatic aneurysms remains a controversial issue. Rubin et al.,5 Labropoulos et al.,6 as well as others,15 recommend close surveillance with duplex scanning for asymptomatic patients. The case of paradoxical embolic stroke due to PVA in association with patent foramen ovale is extremely rare. We found 3 similar cases in the literature, 2 men and 1 woman.16 18 The best treatment modality to prevent recurrent stroke in patients with PFO remains to be defined. There are four major choices: (1) medical therapy with anticoagulants; (2) medical therapy with antiplatelet agents; and (3) surgical or percutaneous
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Fig. 2. Intraoperative views of the popliteal vein aneurysm before (A) and after (B) venorrhaphy.
closure. The Cryptogenic Stroke Study,19 as well as other studies,2 demonstrated that antiplatelet and anticoagulant therapies are of equal benefit in preventing recurrent neurologic events in stroke patients with a PFO. The CLOSURE 1 study was a randomized clinical trial comparing the use of a percutaneously placed PFO occlusive device and best medical therapy versus best medical therapy alone for the prevention of recurrent ischemic neurologic symptoms. This study did not show any difference between medical and surgical treatment.1 In conclusion, when PVA is occurs in the setting of ischemic stroke, paradoxical embolism should be considered. A complete work-up is required and the presence of PFO should be investigated. Surgery is the treatment of choice for the PVA. PFO closure and lifelong anticoagulation are not necessary. Surveillance by Doppler ultrasound is also required.
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