JRSM >
Volume 97, Number 2 >
Pp. 77-78
Department of Surgery, Royal Berkshire Hospital, Reading RG1 5AN, UK
1 Department of Radiology, Royal Berkshire Hospital, Reading RG1 5AN, UK
Correspondence to: Mr R B Galland E-mail: Robert.Galland{at}rbbh-tr.nhs.uk
In cystic adventitial disease, a rare cause of claudication, most patients undergo operative or radiological intervention. There is little evidence to support either approach.
CASE HISTORY
In 1991 a man aged 47 arrived at our casualty department with acute left calf pain. For the previous year he had been experiencing claudication in the left leg after walking about 500 m. Smoking was his only vascular risk factor. On examination he had a cold left foot and no pulses could be felt below the femoral artery on that side. All pulses were present in the right leg. Intravenous digital subtraction angiography showed external compression of the lumen of the left popliteal artery. The vessels in the right leg were normal. MRI (Figure 1) and CT imaging revealed a cystic swelling within the wall of the artery and cystic adventitial disease was diagnosed.
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The next day, without treatment, the foot was warm and the leg was pain-free. At two weeks, claudication distance was nearly 1 km and pulses were normal in both legs. MRI (Figure 2) and CT scanning at five months, when he was still pain-free, showed spontaneous resolution of the cyst. A popliteal ultrasound scan at 10 years confirms the permanent resolution of the cyst and the patient remains symptom-free.
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COMMENT
Adventitial cysts are synovial-like cysts in the adventitial layer of the artery wall. They most commonly affect the popliteal artery but have been reported also in the external iliac, brachial, radial and ulnar arteries. Occasionally they are found in association with saphenous veins in the ankle region. In all cases the diseased artery or vein overlies a joint.
The most common presentation is claudication secondary to popliteal artery stenosis or occlusion. The disease predominantly affects men in the fourth and fifth decades (a male to female ratio of 5 to 1). The aetiology is unclear and is reflected by the changing nomenclature— cystic adventitial degeneration, cystic mucoid degeneration, cystic myxomatous adventitial degeneration, subadventitial pseudocyst and cystic adventitial disease. According to the ganglion theory, adventitial cysts are formed and maintained by communication with a synovial space.1 The embryology theory proposes that mucin-secreting mesenchymal cells from the adjacent joint are included in the adventitia of the artery during development.2
Treatment options are excision of the cyst and preservation of the artery, excision of the diseased artery with interposition grafting, CT or ultrasound guided percutaneous drainage or conservative management. Most experience is with the first two methods. In a review of 155 cases of cystic adventitial disease managed between 1954 and 1955,3 69 were grafted and 68 treated by cyst evacuation and removal of the cyst wall. Both groups had an initial success rate of around 94% but little is known about the long-term outcomes of either. There are no controlled trial data or large single-centre series to show the superiority of any one technique. Long-term patency of vein or synthetic grafts used to bypass diseased artery has not been documented. A 10% recurrence rate has been suggested for cyst wall enucleation.4 There are four reported cases of treatment by percutaneous aspiration. In one of these cases aspiration was not possible because the contents were too viscous;5 in another early recurrence led to definitive surgery;6 and the other two were successfully treated.
Three other cases of spontaneous resolution of cystic adventitial disease of the popliteal artery have been found.7–9 We are unable to explain why their natural history is different from that of most other reported patients. The long-term remission in our case suggests that conservative management is justifiable in some patients with non-critical ischaemia. Even in patients with critical ischaemia percutaneous drainage of the cyst may be worth further study. We know from the present case that symptomatic remission is possible without formal excision of the cyst.
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