Cas cliniques

DOI of or

DepartmenTrust, St. Mar

CorrespondLondon SW20rehman01@im

Ann Vasc SurDOI: 10.1016/� Annals of V�Edit�e par ELS

Un cas de kyste adventiciel ilio-f�emoralr�ecidivant

Syed Rehman, Louise Hancock, John Wolfe, Londres, Angleterre

Nous pr�esentons le cas d’un homme de 39 ans pr�esentant une claudication intermittenter�ecidivante 4 ans apr�es la mise en place d’un patch en Dacron au niveau de l’art�ere f�emoralecommune droite pour un kyste adventiciel. L’angiographie par r�esonance magn�etique n’�etait pascontributive, et l’art�eriographie conventionnelle retrouvait une st�enose �a 90% de l’art�eref�emorale commune droite. Le patient �etait trait�e par excision et remplacement de l’art�ereaffect�ee par une proth�ese en PTFE, et restait asymptomatique �a 6 mois. En cas dekyste adventiciel r�ecidivant, l’excision et le remplacement de l’art�ere affect�ee par interpositionproth�etique permet un r�esultat satisfaisant avec une faible probabilit�e de r�ecidive.

CASE REPORT

A 39-year-old man presented with progressive right leg

claudication that prevented him from walking 4 km to

work. He had cystic adventitial disease of the right

common femoral artery and, apart from a history ofmode-

rate smoking, he had no other risk factors for atheroscle-

rotic disease. Four years earlier, he underwent successful

Dacron patch repair of the right common femoral artery.

The cystic degeneration appeared to affect the anterior

two thirds of the artery, which was removed and replaced

with a large Dacron patch. The macroscopic disease was

removed. He remained asymptomatic for 4 years before

presenting with a recurrence of his symptoms.

Duplex ultrasound showed that the luminal diameter

of the right common femoral artery had narrowed,

although therewere reasonable velocities passing through

it and treadmill Doppler test was positive. Magnetic reso-

nance angiography (Fig. 1) showed signal loss in the distal

right common femoral artery and proximal superficial

femoral artery, indicating a degree of turbulence, but it

iginal article: 10.1016/j.avsg.2009.05.020.

t of Vascular Surgery, Imperial College Healthcare NHSy’s Hospital, Londres, Angleterre.

ence : Syed Rehman, 87 Grand Drive, Raynes Park,9DW, United Kingdom, E-mail addresses: syed.

perial.ac.uk, syedrehman@o2email.co.uk

g 2010; 24: 550.e1-550.e3j.acvfr.2010.12.008ascular Surgery Inc.EVIER MASSON SAS

was multiplanar angiography (Fig. 2) that identified

extensive cystic adventitial disease of the posterior wall

causing 90% stenosis of the right common femoral artery

and origin of the right superficial femoral artery. The

patient’s worsening symptoms were considered to be due

to either aggressive myointimal hyperplasia or recurrence

of the cystic adventitial disease affecting the remaining

part of the common femoral artery that was previously

unaffected.

On the basis of the patient’s symptoms and the results

of the investigations, it was decided that it would be

appropriate to excise and replace the affected length of

common femoral artery. Intraoperatively, a large cystic

sac within the adventitia was compressing the right

common femoral artery and the origin of the right

superficial femoral artery. The termination of the external

iliac artery was transected, the profunda femoris was

transected just beyond its origin, and the superficial

femoral artery was transected 4 cm beyond its origin. An

8-mm PTFE externally supported graft was then anasto-

mosed to the external iliac artery end-to-end and the

distal anastomosis was performed end-to-end to the pro-

funda femoris artery. Following this, flow was restored to

the profunda femoris. An anastomosis was then per-

formed to the superficial femoral artery end-to-end and

the proximal anastomosis was end-to-side onto the PTFE

graft. Excellent flows were achieved as a result. Histology

revealed that the resection margins were free of cystic

adventitial disease. At 6-month follow-up, the patient’s

symptoms had completely resolved, he had a full com-

plement of peripheral pulses in the right leg, and treadmill

Doppler results were normal.

600.e1

Fig. 1. Magnetic resonance angiogram that is incon-

clusive for stenosis of the right common and superficial

femoral arteries.

Fig. 2. Multiplanar computed tomography angiogram

demonstrating severe stenosis of the right common

femoral artery and origin of the right superficial femoral

artery.

600.e2 Cas cliniques Annales de chirurgie vasculaire

DISCUSSION

Cystic adventitial disease is a rare cause of intermit-

tent claudication with an incidence of 1 : 1200 and

predominantly affecting males (male-to-female

ratio 15 : 1).1 The condition was first described by

Atkins and Key2 in 1947 involving the external iliac

artery in a 40-year-old man. Since then, approxi-

mately 320 cases have been reported.1 The majority

of these cases have involved the popliteal artery;

only 36 cases report iliofemoral artery

involvement.1,3

The etiology of this condition remains unclear.

Several theories have been suggested and were

most recently reviewed by Levien and Benn in

1998.4 These include the suggestions that cystic

adventitial disease is part of a myxomatous systemic

degenerative condition, caused by repeated micro-

trauma; the ganglion theory that adventitial cysts

originate as capsular synovial structures that involve

the adjacent blood vessel; and the developmental

theory that mucin-secreting cells from an adjacent

joint become included in the adventitia during

development. Levien and Benn suggested their own

unifying hypothesis that cystic adventitial disease is

a developmental condition caused by nonsynovial

less-differentiated joint-related mesenchymal cell

rests in nonaxial blood vessels. Cystic adventitial

disease should be suspected in patients with

intermittent claudication in the absence of peri-

pheral arterial disease or atherosclerotic risk factors.

It may present acutely but does not usually manifest

as rest pain. Clinically, there are diminished or

absent pulses, and a bruit may be audible over the

stenosed artery. The ankle-brachial pressure index

may also be reduced.

Investigations include duplex ultrasound, which

shows arterial stenosis and a cyst, with no flow,

appearing as anechoic or hypoechoic masses in the

vessel wall. Angiography demonstrates distinctive

stenosis of the arterial lumen, which has an ‘‘hour-

glass’’ appearance or demonstrates the ‘‘scimitar

sign’’ depending on the configuration of the cyst.

Magnetic resonance imaging (MRI) is said to be as

effective at diagnosing cystic adventitial disease as

conventional angiography. Cysts are hyperintense

on T2-weighted images and have variable signal

intensity on T1-weighted images due to the variable

amount of mucoid material within the cysts.5

However, in our patient, MRI (Fig. 1) was incon-

clusive, whereas multiplanar angiography (Fig. 2)

defined the severe stenosis.

Several treatment methods have been used in the

management of cystic adventitial disease. In a small

number of cases, conservative management has

resulted in spontaneous resolution of symptoms.6-9

However, the majority of patients require interven-

tion. Ultrasound- or computed tomography (CT)-

guided aspirationhas beenusedwith varying success.

Do et al.10 describe successful ultrasound-guided

Vol. 24, No. 4, 2010 Cas cliniques 600.e3

aspiration in seven patients without recurrence at a

mean follow-up of 14.8 months, while Sieunarine

et al.11 and Colombier et al.12 both report recurrences

following CT-guided aspiration requiring subsequent

surgical intervention. Our experience suggests that

recurrence is likely unless the segment of artery is

removed. Balloon angioplasty does not appear to be

successful as it does not affect the cystic compression

of the artery.13

Surgical intervention appears to provide the

most successful long-term outcome, which is of

particular relevance as cystic adventitial disease

affects relatively young patients. Cyst excision can

be performed without arterial reconstruction but

is associated with a 10% recurrence rate.14 We

have reported recurrence in a patient who origi-

nally had cyst excision performed with patch

repair. Ohta et al.15 reported a case of cystic

adventitial disease recurring within an interposed

autogenous saphenous vein graft.

In patients with recurrent cystic adventitial

disease, excision and replacement of the affected

artery with a prosthetic interposition graft provides

a successful outcome with minimum chance of

recurrence. However, it is still important to consider

the complications of prosthetic graft interposition.

In addition, it is vital that all patients receive

follow-up.

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