Case Report
Cystic adventitial disease of the popliteal artery: Two case reports and a review of the literature
Vascular 2015, Vol. 23(2) 204–210 ! The Author(s) 2014 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/1708538114541652 vas.sagepub.com
P Del Canto Peruyera, MJ Vallina-Victorero Va´zquez, ´ lvarez, A A ´ lvarez Salgado, M Botas Velasco, P Calvı´n A ´ ´ J Cervin˜o Alvarez and LJ Alvarez Ferna´ndez
Abstract Two cases of cystic adventitial disease treated at our institution over the last year are presented. They were middle-aged and apparently healthy patients, and the symptoms begin with a sudden onset of unilateral claudication. After performing a magnetic resonance angiography, a cystic formation attached to the adventitia of the popliteal artery was identified. Both patients were treated in the same manner, with resection of the affected arterial segment and vein bypass interposition. Both remain asymptomatic after one year of follow-up in one case and six months in the other. Cystic adventitial disease is a rare entity, which presents in patients without cardiovascular risk factors, so sometimes it takes long to reach a definitive diagnosis. Concerning the different treatment options, cyst excision together with the affected arterial segment seems to offer better mid- and long-term results when compared with other treatment options such as cyst aspiration or endovascular techniques, although there are no multicenter trials evidencing the superiority of one against the others.
Keywords Cystic adventitial disease, popliteal artery, diagnosis, treatment
Introduction Cystic adventitial disease (CAD) is a rare vascular disease of unknown etiology which results in unilateral intermittent claudication in young patients without classic cardiovascular risk factors.1 Diagnosis is made from imaging tests such as ultrasound, computed tomography (CT) angiography (CTA), or magnetic resonance angiography (MRA), the latter being the diagnostic modality that offers more information.2 Regarding the treatment, multiple techniques are described, the open surgical management being the one which count on the greatest acceptance.
absence of distal pulse and an ankle-brachial index (ABI) of 0.76; in the contralateral limb, all pulses are present with an ABI of 1.25. With all these data, we suspect an embolic event already compensated, the reason why we start anticoagulant therapy with low-molecular-weight heparins prior to oral anticoagulation. One month later, the patient returns with clinical worsening. The patient maintains femoral pulse but has lost popliteal pulse (which was present during the first consultation); in the same way, ABI has fallen to 0.30.
Department of Angiology, Vascular and Endovascular Surgery, Cabuen˜es Hospital, Gijo´n, Spain
Case report 1 A 54-year-old man, with paroxysmal atrial fibrillation as sole cardiovascular risk factor, presents to our consultations, with a sudden onset of left calf claudication at 100 m of six-week duration. Left lower limb (LLL) presents normal femoral and popliteal pulses, with
Corresponding author: P Del Canto Peruyera, Department of Angiology, Vascular and Endovascular Surgery, Cabuen˜es Hospital, Camino de los Prados 395, Gijo´n 33394, Spain. Email: [email protected]
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Suspecting a new embolic event, we decided to perform a percutaneous arteriography, in which we note that all arteries (iliac, femoral, popliteal, and distal vessels) are patent and without lesions (Figure 1). After arteriography, the patient recovers popliteal and distal pulses spontaneously, without clinical claudication, so hospital discharge is authorized. Four days after hospital discharge, the patient presents with rest pain in LLL. Physical examinations have changed once again, with a femoropopliteal obstruction and an ABI of 0.40. We decided patient admission and initiated treatment with intravenous E1 prostaglandins, waiting for CTA performing. This CTA is performed two days later and shows an occlusion in the mid portion of the popliteal artery, without lesions at the other arterial territories. In view of the CTA findings, we perform an embolectomy. Fogarty catheter progresses till the foot, providing resistance to the passage of the catheter through the mid portion of the popliteal artery, but no thrombus could be extracted. During the same procedure, we perform a control arteriography which shows a 2-cm length occlusion at the popliteal artery (Figure 2). As the surgical procedure is being carried out under local anesthesia, we decide to conclude the surgical intervention, and then perform an MRA in the next few days which can provide more information about the case, to try to reach a definitive diagnosis. It is in this MRA where we note the presence of the
cystic formation at the popliteal artery, which explains the occlusion that the patient presents at this level (Figure 3). Once diagnosed, and although the patient recovers popliteal and distal pulses again, we offer him to perform a surgical intervention for a complete resolution of the problem. The patient accepts, and the surgical intervention is carried out through a posterior approach, with a cyst excision together with the affected arterial segment (Figure 4), for later to perform an end-to-end bypass using the ipsilateral lesser saphenous vein. Both the procedure and the immediate postoperative period elapse without complications; the patient recovers popliteal and distal pulses and continues asymptomatic after one-year follow-up.
Figure 1. First arteriography performed which shows that all the arteries are patent and without lesions.
Figure 2. Arteriography showing an occlusion at the popliteal artery.
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Case report 2 A 49-year-old woman presents to the consultation with left calf claudication at 300 m of one-year duration; she presents hypercholesterolemia as the only cardiovascular risk factor. LLL presents normal femoral and popliteal pulses, with absence of distal pulses, and the contralateral limb has normal pulses at all levels, with an ABI of 0.70 and 1.10, respectively.
We add acetylsalicylic acid (ASA) at dose of 100 mg/ day to her previous treatment, and we make an appointment within three months. When the patient is reevaluated, she complains about clinical worsening; there are no changes in physical examination, although the ABI is now of 0.60. An MRA is performed on an outpatient, which shows as in the previous case a cystic formation at the adventitia tunica in the mid portion of the popliteal artery. Because the patient is unable to very basic activities of daily living, we offer her the same surgical intervention that was done with the previous patient. She accepts surgical management and as in the first case, both the procedure and the immediate postoperative period elapse without complications, recovering popliteal and distal pulses. After six-month follow-up, she continues asymptomatic.
Discussion
Figure 3. MRA where the presence of the cystic formation at the popliteal artery is noted first.
Figure 4. Affected popliteal artery segment in which we observe the arterial lumen collapsed by de cystic formation at the tunica adventitia.
CAD is a rare disease, with an estimated incidence of
Cystic adventitial disease of the popliteal artery: Two case reports and a review of the literature
Vascular 2015, Vol. 23(2) 204–210 ! The Author(s) 2014 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/1708538114541652 vas.sagepub.com
P Del Canto Peruyera, MJ Vallina-Victorero Va´zquez, ´ lvarez, A A ´ lvarez Salgado, M Botas Velasco, P Calvı´n A ´ ´ J Cervin˜o Alvarez and LJ Alvarez Ferna´ndez
Abstract Two cases of cystic adventitial disease treated at our institution over the last year are presented. They were middle-aged and apparently healthy patients, and the symptoms begin with a sudden onset of unilateral claudication. After performing a magnetic resonance angiography, a cystic formation attached to the adventitia of the popliteal artery was identified. Both patients were treated in the same manner, with resection of the affected arterial segment and vein bypass interposition. Both remain asymptomatic after one year of follow-up in one case and six months in the other. Cystic adventitial disease is a rare entity, which presents in patients without cardiovascular risk factors, so sometimes it takes long to reach a definitive diagnosis. Concerning the different treatment options, cyst excision together with the affected arterial segment seems to offer better mid- and long-term results when compared with other treatment options such as cyst aspiration or endovascular techniques, although there are no multicenter trials evidencing the superiority of one against the others.
Keywords Cystic adventitial disease, popliteal artery, diagnosis, treatment
Introduction Cystic adventitial disease (CAD) is a rare vascular disease of unknown etiology which results in unilateral intermittent claudication in young patients without classic cardiovascular risk factors.1 Diagnosis is made from imaging tests such as ultrasound, computed tomography (CT) angiography (CTA), or magnetic resonance angiography (MRA), the latter being the diagnostic modality that offers more information.2 Regarding the treatment, multiple techniques are described, the open surgical management being the one which count on the greatest acceptance.
absence of distal pulse and an ankle-brachial index (ABI) of 0.76; in the contralateral limb, all pulses are present with an ABI of 1.25. With all these data, we suspect an embolic event already compensated, the reason why we start anticoagulant therapy with low-molecular-weight heparins prior to oral anticoagulation. One month later, the patient returns with clinical worsening. The patient maintains femoral pulse but has lost popliteal pulse (which was present during the first consultation); in the same way, ABI has fallen to 0.30.
Department of Angiology, Vascular and Endovascular Surgery, Cabuen˜es Hospital, Gijo´n, Spain
Case report 1 A 54-year-old man, with paroxysmal atrial fibrillation as sole cardiovascular risk factor, presents to our consultations, with a sudden onset of left calf claudication at 100 m of six-week duration. Left lower limb (LLL) presents normal femoral and popliteal pulses, with
Corresponding author: P Del Canto Peruyera, Department of Angiology, Vascular and Endovascular Surgery, Cabuen˜es Hospital, Camino de los Prados 395, Gijo´n 33394, Spain. Email: [email protected]
Downloaded from vas.sagepub.com by guest on November 14, 2015
Peruyera et al.
205
Suspecting a new embolic event, we decided to perform a percutaneous arteriography, in which we note that all arteries (iliac, femoral, popliteal, and distal vessels) are patent and without lesions (Figure 1). After arteriography, the patient recovers popliteal and distal pulses spontaneously, without clinical claudication, so hospital discharge is authorized. Four days after hospital discharge, the patient presents with rest pain in LLL. Physical examinations have changed once again, with a femoropopliteal obstruction and an ABI of 0.40. We decided patient admission and initiated treatment with intravenous E1 prostaglandins, waiting for CTA performing. This CTA is performed two days later and shows an occlusion in the mid portion of the popliteal artery, without lesions at the other arterial territories. In view of the CTA findings, we perform an embolectomy. Fogarty catheter progresses till the foot, providing resistance to the passage of the catheter through the mid portion of the popliteal artery, but no thrombus could be extracted. During the same procedure, we perform a control arteriography which shows a 2-cm length occlusion at the popliteal artery (Figure 2). As the surgical procedure is being carried out under local anesthesia, we decide to conclude the surgical intervention, and then perform an MRA in the next few days which can provide more information about the case, to try to reach a definitive diagnosis. It is in this MRA where we note the presence of the
cystic formation at the popliteal artery, which explains the occlusion that the patient presents at this level (Figure 3). Once diagnosed, and although the patient recovers popliteal and distal pulses again, we offer him to perform a surgical intervention for a complete resolution of the problem. The patient accepts, and the surgical intervention is carried out through a posterior approach, with a cyst excision together with the affected arterial segment (Figure 4), for later to perform an end-to-end bypass using the ipsilateral lesser saphenous vein. Both the procedure and the immediate postoperative period elapse without complications; the patient recovers popliteal and distal pulses and continues asymptomatic after one-year follow-up.
Figure 1. First arteriography performed which shows that all the arteries are patent and without lesions.
Figure 2. Arteriography showing an occlusion at the popliteal artery.
Downloaded from vas.sagepub.com by guest on November 14, 2015
206
Vascular 23(2)
Case report 2 A 49-year-old woman presents to the consultation with left calf claudication at 300 m of one-year duration; she presents hypercholesterolemia as the only cardiovascular risk factor. LLL presents normal femoral and popliteal pulses, with absence of distal pulses, and the contralateral limb has normal pulses at all levels, with an ABI of 0.70 and 1.10, respectively.
We add acetylsalicylic acid (ASA) at dose of 100 mg/ day to her previous treatment, and we make an appointment within three months. When the patient is reevaluated, she complains about clinical worsening; there are no changes in physical examination, although the ABI is now of 0.60. An MRA is performed on an outpatient, which shows as in the previous case a cystic formation at the adventitia tunica in the mid portion of the popliteal artery. Because the patient is unable to very basic activities of daily living, we offer her the same surgical intervention that was done with the previous patient. She accepts surgical management and as in the first case, both the procedure and the immediate postoperative period elapse without complications, recovering popliteal and distal pulses. After six-month follow-up, she continues asymptomatic.
Discussion
Figure 3. MRA where the presence of the cystic formation at the popliteal artery is noted first.
Figure 4. Affected popliteal artery segment in which we observe the arterial lumen collapsed by de cystic formation at the tunica adventitia.
CAD is a rare disease, with an estimated incidence of
