Cystic
disease of the popliteal artery
(Portuguese
PDF version)
Douglas
Faria Corrêa Anjo, German Marcelo Negrão Gimenez, Daniela
Belia da Silva*
*
Hospital e Maternidade Jaraguá, Jaraguá do Sul, SC,
Brazil.
Correspondence:
Douglas Faria Corrêa Anjo
Rua Guilherme Cristiano Wackerhagen, 405, Vila Nova
CEP 89259-300 - Jaraguá do Sul, SC
Brazil
ABSTRACT
Cystic
disease of the popliteal artery is a rare but well recognized cause
of intermittent claudication. The etiology of the disease is still
controversial and the literature reports various hypotheses for
its origin. Diagnosis starts with history and physical examination;
diagnostic studies comprise color duplex scan, digital angiography
and magnetic resonance imaging. A 69-year-old female presented with
intense burning in her leg and intermittent claudication for 50
m, for a long period of evolution, entered this study. The exams
revealed the presence of a cystic structure in the popliteal artery
causing complete occlusion of its lumen. Surgery was performed with
cystic resection and we preferred no to excise the affected segment
from the popliteal artery.
Key
words: popliteal artery, cysts, intermittent claudication.
Palavras-chave: artéria poplítea,
cistos, claudicação intermitente.
J
Vasc Br 2004;3(3):277-80
Adventitial
cystic disease of the popliteal artery is a rare entity which should
not be overlooked in the examination of patients with intermittent claudication.
It usually affects young patients, but may also occur in elderly individuals,
as in the case reported herein. The disorder is characterized by the
development of mucin-containing cysts inside the adventitia, causing
localized stenosis or occlusion of the arterial lumen. Its etiopathogenesis
remains obscure, despite the existence of several theories, for example
that which states that the presence of repeated microtrauma causes rupture
and subsequent cystic degeneration of the adventitia. Nishibe et al.1
also describe three additional possibilities: myxoid degeneration, ganglionar
involvement of adjacent vascular structures and (most widely accepted)
the inclusion of remnants of mucus-secreting synovial cells in the adventitia
in the popliteal artery adventitia.
The main objective of this paper is to underscore the importance of
non-invasive methods in the diagnosis of vascular lesions of the lower
extremities.
CASE
REPORT
We report
the case of a 69-year old woman complaining of intermittent claudication
for a 50 m range and intense burning pain below the knee. The patient
was a smoker and presented hypertension. The routine laboratory tests
did not reveal changes in glycemia or lipid levels. She was at that
moment using imipramide, omeprazole and pentoxiphyllin. On physical
examination, all pulses were present, without visible or palpable tumors
in the popliteal fossa. A Doppler scan, however, did not disclose any
trophic lesions or edema. The ankle/brachial pressure index was about
0.5.
A Doppler scan (Figure 1) revealed that both the common femoral artery
and the superficial femoral artery were patent, with triphasic spectrum
of laminar flow. However, a slight thickening of the arterial intima
and calcification points were also observed. A cystic lesion was observed
anteriorly in the popliteal artery, measuring 17 x 36 mm. The lesion
was compressing the artery, causing a reduction in distal arterial flow.
Digital angiography was also performed, showing segmental occlusion
of the popliteal artery (Figure 2), with rich collateral circulation
filling the leg arteries up to the arch of the foot. Two remarkable
features were the absence of atherosclerotic lesions up and downstream
from the occlusion and the narrowness of the contrast filament in the
artery wall. We chose to perform a nuclear magnetic resonance examination,
which confirmed the existence of a popliteal cyst compressing the popliteal
artery and vein, in addition to the nerve (Figure 3).
 Figure
1 -Doppler scan: presence of cyst compressing the popliteal artery.
Figure
2 - Digital angiography. Segmental occlusion. Note the presence of collateral
circulation and the normal appearance of the artery distally from the
cyst.
Figure
3 - Magnetic resonance angiography. Note the exact location of the cyst.
The indication
was for surgical treatment. With the patient lying on her stomach, a
classic incision (medial or posterior?)
was performed for approaching the popliteal artery. The artery was identified
and tied proximally and distally. We then began dissecting the cyst,
which was firmly adhered to the artery, changing its natural anatomic
course. This forced us to incise the tumor to facilitate its dissection.
After opening the cyst and removing a great amount of mucinous material,
it was possible to separate the cyst from the artery. At that moment
the return of a strong pulse was observed, which had been undetectable
during dissection. There was also an important increase in caliber along
the entire artery. At this point, transoperative angiography revealed
the filling of previously occluded segments and the disappearance of
collateral circulation (Figure 4). Despite the presence of filling defects,
and faced with the dilemma of either interposing a saphenous vein graft
or wait and observe, we decided to finalize the surgical procedure and
follow the case clinically (Figure 5).
Figure
4 - Transoperative angiography. Filling defects are observed. Absence
of collateral circulation.
Figure
5 - Surgical aspect after the artery was released from compression.
The tweezers indicate the compression site.
The material
excised during the surgery was submitted to anatomic and pathological
analysis which revealed the presence of a fibrous capsule. However,
the technical conditions of our laboratory did not allow the contents
of this capsule to be evaluated. The patient was discharged on the second
postoperative day, without complaints. She was advised to resume regular
walking, avoid smoking and take a platelet-blocking drug (200 mg).
A Doppler scan performed 10 days after the surgery showed a patent popliteal artery and vein, without the presence of thrombi. The patient remained free of symptoms after 1 year of follow-up.
DISCUSSION
Adventitial
disease is a rare entity, and there are no large series described in
the literature. In Brazil, a series of five cases was described by Luz
et al. 2 in 1990. Reports such as that of
Rollo et al.3 confirm the rarity of this
condition. In Argentina, Abelleyra et al.,4
reviewing 579 cases submitted to surgery due to diseases involving the
popliteal artery, found two cases of cystic disease. Similarly, in 2003
Rispoli et al.5 reported on one case of
adventitial cystic disease of the popliteal artery.
In the
present case, intermittent claudication was not the patient's main complaint,
despite the limited 50-m walking range. Rather, she was mostly bothered
by the intense burning that prevented her from sleeping, especially
when the knee was bent. She had suffered the symptom for years, and
had always been diagnosed with "rheumatism," which leads us
to believe that the development of the disease was very slow.
We made a point of approaching the cyst during the surgical procedure,
since we were convinced that the construction of a medial bypass alone
would not solve the problem, since it would not be sufficient to release
the compression of the vein and nerve. It should be underscored that
the entire arterial tree, up and downstream from the injured site was
free of hemodinamically significant processes, despite the patient's
age. This is very important, since the existence of other stenosed areas
could have masked the problem and also prevent the cyst from being identified,
which could have led to inadequate treatment.
Concerning diagnosis, in addition to the patient's history and to the
detailed physical exam, the sequence we employed, starting with a Doppler
scan, seems to be the most indicated for identifying adventitial cystic
disease of the popliteal artery. This is corroborated in the work of
Elias et al.,6 who describe a similar case,
with practically matching arteriography and magnetic resonance angiography
results. Angiography is especially useful to provide details concerning
the morphology of the arterial wall, arterial contents and aspects of
the adjacent musculoskeletal structures, as well as about the circulation
status. It was an extremely important tool in the present case, since
it allowed us to rule out the presence of atherosclerotic lesions. However,
the Doppler scan is currently the method of choice to start the diagnostic
investigation, because it is non-invasive and provides a correct diagnosis,
in addition to being widely available at a lower cost in comparison
to other preliminary exams.
The treatment of adventitial cystic disease of the popliteal artery
is surgical. In many publications, the treatment reported consisted
of the resection and replacement of the injured segment, preferably
with a venous graft. In the present case, however, the surgical correction
did not require arterial resection. Since we observed the return of
a strong arterial pulse, we chose to simple resect the cyst. This choice
was also referred by Ishikawa,7 when reporting
on 80 cases in 1987, and mentioned by Castiglia.8
In other cases, the only efficient alternative was aspiration of the
cyst. Until the present moment, there are no reports of endovascular
treatment of cystic disease of the popliteal artery.
Our main objective in reporting this case was to call attention to adventitial
cystic disease of the popliteal artery as a diagnostic possibility when
examining patients with intermittent claudication, and also to underscore
the importance of Doppler scans. Although the disease affects mostly
young adults, a slow-developing cyst may produce symptoms only at a
more advance age, complicating diagnosis and the establishment of adequate
treatment.
REFERENCES
1.
Nishibe M, Nishibe T, Yamasshita T, Kato H, Kudo F, Yasuda K. Cystic
adventitial disease of popliteal artery: etiologic considerations. J.
Cardiovasc Surg (Torino) 2002;43(4):573-4.
2. Luz N, Vieira G, Bertencini R, Ceola, L. Cistos poplíteos
e angiopatias. ACM Arq Catarin Med. 1990;19:273-6.
3.Rollo
HA, Gama JC, Lastoria S, Yoshida W, Maffei FH. Cisto de adventícia
em artéria poplítea. Relato de dois casos. AMB (Associação
Médica Brasileira)1982;289:79-81.
4.
Abelleyra J, Grandjean M, Letrenta S, Chikiar D. Patologia de la artéria
poplítea. Rev Argent Cri 2001;80(5):171-9..
5. Rispoli P, Moniaci D, Zan S, et al. Cystic adventitial
disease of popliteal artery. Report of one case and review of the literature.
J Cardiovasc Surg (Torino). 2003;44(2):225-8.
6.
Elias DA, White LM, Rubenstein JD, Chritakis M, Merchant N. Clinical
evaluation and MR imaging of popliteal artery entrapment and cystic
adventitial disease. AJR Am Roentgenol 2003;180(3):627-32.
7.
Ishikawa K. Cystic adventitial disease of the popliteal artery and other
stem vessels in the extremities. Jpn J Surgery 1987;17:221.
8. Castigilia V. Doença cística das artérias.
In: Maffei FHA. Doenças Vasculares Periféricas. 2ª
ed. Rio de Janeiro: Medsi; 1995. p.691-700.
|
