ARTICLE
Auteur(s) :, Roeland CEULEN1,*, Yolande
BULLENS-GOESSENS1, Michiel DE HAAN2, Joep
VERAART1
1Department of Dermatology University Hospital
Maastricht PO Box 5800, 6202 AZ Maastricht The Netherlands
2Department of Radiology University Hospital Maastricht
PO Box 5800, 6202 AZ Maastricht The Netherlands
*R.P.M. Ceulen. Fax: (+31) 43 38 772 93.
E-mail: Rce@sder.azm.nl
accepté le 16 Mars 2004
Venous leg ulceration is a medical challenge that consumes up to 1%
of the national health care resources in the Western world [1]. The
incidence of severe CVI is 6-8% and the prevalence of venous
ulceration is 1% in the population [2-4]. The incidence of venous
leg ulceration increases with age, although it can occur much
earlier in life [1]. In the majority of these cases varicose veins
are not always the underlying cause and therefore further
investigation is recommended. We want to illustrate this in the
following case report.
Clinical observations
A 45-year-old female was referred to our hospital, having had a
painful and therapy resistant ulcer on the medial malleolus of the
left lower leg for 8 months. She was treated with antibiotic
ointment and non-elastic compression bandages. The initial cause of
the ulceration was not known. She was known to have had an isolated
deep venous thrombosis of the external iliac vein of the left leg
that had occurred 10 years ago after knee surgery. She was
healthy and did not use anticoagulants, nor any other drugs.
On the left medial malleolus there was a 4-cm2
non-granulating ulcer, covered with a yellow cuff of fibrin
surrounded by superficial erosion (( Fig. 1 )). Except from
a superficial spontaneous De Palma vein, no other signs of venous
varicosity or venous insufficiency were seen. The physical
examination and laboratory tests were normal. Blood was drawn for
antithrombin, protein C and S, factor V Leiden mutation, lupus
coagulant and cardiolipines, which were normal.
Duplex ultrasound of the left leg showed residual thrombus and
reflux in the superficial femoral vein. Side branches, including
the epigastric and the De Palma vein showed spontaneous flow. The
superficial and deep venous system showed a normal venous flow. A
perforating vein arising from the posterior tibial vein showed
reflux. Ascending phlebography indicated residual signs of an old
thrombus in the external iliac vein and a diminished filling of the
pelvic and collateral veins (( Fig. 2 )).
Because of the narrow lumen of the left external iliac vein,
balloon dilation was performed (Diamond Back 8/40 mm, Boston
Scientific), followed by stent placement (Wallstent 10/68 mm,
Boston Scientific). After this, the flow through the collateral
veins diminished immediately (( Fig. 3 )). The patient
was anticoagulated with acenocoumarol for 3 months. During a
4-month period the ulcer healed slowly but completely. During a
12-month follow-up period no recurrences were seen.
Discussion
We described a patient with a therapy-resistant and spontaneous
recurrent venous leg ulcer due to partial occlusion of the iliac
vein. In 45-60% of all leg ulcers venous disease is the primary
cause [5]. Fifty percent of all patients with leg ulcers have a
history of deep venous thrombosis (DVT) [6]. The incidence rate of
isolated iliac vein thrombosis is 0.26% [7]. Iliac vein occlusions
can be caused by thrombosis, or less commonly by trauma or
irradiation, or as a result of external compression of deep veins
by retroperitoneal fibrosis and tumours. They also can be
iatrogenic after an operative injury or after compression of the
left common iliac vein by the overlying right common iliac artery
(May-Thurner syndrome) [8-10].
A common and often disabling consequence of DVT is the post
thrombotic syndrome (PTS) which occurs in 60% of the patients
within 2 years. Symptoms of PTS range from mild oedema to
incapacitating swelling with pain and ulceration [11]. The
frequency of isolated iliac or femoral thrombosis is almost 22%
[12]. Although most patients with chronic post thrombotic pelvic
venous obstruction have symptoms known as venous claudication
(pain, tense swelling and cyanosis), 25% do not have those symptoms
[13, 14].
When symptoms of a previous venous thrombosis have been
recognised or suspected, the underlying aetiology is rarely
immediately clear. Proper evaluation of venous system diseases
often requires both functional and anatomic information about the
venous circulation. Several important functional tests proved to be
useful in helping to assess the condition of the superficial and
deep venous systems.
Ascending phlebography is used to visualise the entire venous
tree from ankle to the inferior vena cava. It identifies obstructed
vein segments and most of the recanalised post-thrombotic segments.
Descending phlebography makes clear whether the valves are
competent or not. For a long time contrast phlebography was
considered to be the gold standard. The disadvantages of
phlebography (radiation, dye exposure, costs) have led to the
development of non-invasive techniques [15]. Nowadays symptomatic
patients undergo phlebography only if there is incongruity between
physical examination, history, and non-invasive studies.
Duplex ultra sonography and functional venous outflow
plethysmography are used to confirm vein occlusion. It is important
to exclude any abdominal or pelvic pathology with computed
tomography or magnetic resonance imaging. The non-invasive duplex
investigation, today’s gold standard, allows direct visualisation
of the vein and identification of flow through venous vessels [16].
For chronic deep vein thrombosis, the sensitivity and specificity
for venous duplex imaging are respectively 75 and 86% [17]. In
chronic iliac vein thrombosis however, duplex data could be
unreliable because of poor visualisation in the upper pelvis, since
the common iliac vein is occluded [18].
Venous outflow plethysmography is used to determine quantitative
venous function. The venous filling index and the calf muscle pump
ejection fraction are useful, but can only regionally detect
significant reflux. With a sensitivity of 71% and a specificity of
83%, air plethysmography can produce misleading data of the chronic
pelvic vein obstruction [15]. The outflow fraction can be normal
due to collateral veins and the degree of valvular reflux can
become unclear because of a low flow state. Therefore it is useful
to perform hemodynamic tests to identify and separate superficial
and deep vein reflux, which must be distinguished from obstruction.
In order to determine the significance of obstruction to venous
flow direct femoral vein pressure should be measured [19, 20].
In conclusion we like to propose that, although rare, the
existence of pelvic vein thrombosis and obstruction can be a cause
of venous leg ulceration. Because standard non-invasive
duplex-ultrasound investigation can fail to demonstrate the
obstruction, especially in the pelvic veins, invasive phlebography
should be performed in suspected cases as a secondary test. Since
duplex scanning is known as the gold standard, air-pletysmography
and hemodynamic tests are not routinely proposed. Therapeutic
option is percutaneous iliac venous stenting, which is a safe and
efficient method to correct pelvic venous outflow obstruction.
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