Case Report
Lipiodol-based Lymphangiography and Glue-based Embolization of Retroperitoneal Lymphatic Vessels to Treat Symptomatic Retroperitoneal Lymphocele: A Case Report
Nariman Nezami, Haddy Jarmakani, Angelo Marino, Todd Schlachter, Igor Latich, Rajasekhara Ayyagari and Juan Carlos Perez Lozada*
Department of Radiology and Biomedical Imaging, Yale School of Medicine, USA
*Corresponding author: Juan Carlos Perez Lozada, Department of Radiology and Biomedical Imaging, Yale School of Medicine, USA
Published: 01 Oct, 2018
Cite this article as: Nezami N, Jarmakani H, Marino A,
Schlachter T, Latich I, Ayyagari R, et
al. Lipiodol-based Lymphangiography
and Glue-based Embolization of
Retroperitoneal Lymphatic Vessels to
Treat Symptomatic Retroperitoneal
Lymphocele: A Case Report. Clin Surg.
2018; 3: 2131.
Abstract
Lymphocele occur due to the disruption of lymphatic vessels typically in the setting of trauma or
surgery. A number of procedures have been introduced to treat this entity, but none have been
entirely successful. Here, we present a 67-year-old female diagnosed with stage III ovarian cancer
who developed a retroperitoneal lymphocele after robotic hysterectomy, bilateral salpingooophorectomy,
and tumor debulking. She had multiple unsuccessful sessions of sclerotherapy
over two months. Eventually, the lymphatic vessels supplying the lymphocele were mapped
by lipiodol injection in the inguinal lymph nodes and embolized withN-butyl-2-cyanoacrylate
glue. This resulted in resolution of the lymphocele and symptoms. Lipiodol-based mapping and
glue embolization of retroperitoneal lymphatic vessels is a safe and feasible procedure to treat
retroperitoneal lymphoceles.
Keywords: Lymphocele; Retroperitoneal; Embolization; Glue; Lymph vessel; Lipiodol
Introduction
Lymphocele is a known rare complication of surgery or consequence of trauma, caused
by disruption of lymphatic vessels. It mainly occurs after renal transplantation, vascular, spinal,
urologic or gynecologic surgeries [1-6]. Most patients are either diagnosed incidentally on postsurgical
follow up imaging or present with secondary symptoms due to mass effect. Although
surgical resection was initially considered the gold standard treatment for lymphocele [7], its
association with high rate of recurrence, infection, and need for hospitalization quickly replaced the
open procedure with laparoscopic approach [8-10].
In recent years, minimally invasive procedures ranging from simple aspiration, catheterbased
drainage, and transcatheter sclerotherapy have become popular. Simple aspiration alone is
frequently inadequate [8,11]. However, a combination of catheter-based drainage and transcatheter
sclerotherapy produce results comparable to surgery [12]. Sclerotherapy causes wall irritation by
inducing local inflammation and fibrosis of the lymphatic channels, thus obliterating the lymphatic
leak [11]. While there is abundant literature on surgical and trans-catheter approaches, there are
only a few reported cases of glue embolization of pelvic lymphocele. Our case demonstrates a post
pelvic surgery retroperitoneal lymphocele treated with lipiodol mapping and glue embolization of
the feeding lymphatic vessels.
Case Presentation
A 67-year-old female with past medical history of recently diagnosed stage III papillary
ovarian cancer, who is status post adjuvant chemotherapy and robotic assisted hysterectomy,
bilateral salpingo-oophorectomy, tumor debulking with Omentectomy. A month after surgery,
ultrasonography of the retro peritoneum performed for left sided pelvic/back pain showed new
moderate hydronephrosis of the left kidney without visualization of the left ureterovesicaljet, while
free fluid within the pelvis appeared less prominent compared to the ultrasound done prior surgery
(Figure 1). Contrast-enhanced CT scan of abdomen and pelvis five days later revealed significant
left-sided hydroureteronephrosis involving the proximal third of the left ureter, secondary to mass
effect from a large 5.6 cm cystic retroperitoneal hypoattenuating collection, most consistent with a
retroperitoneal lymphocele (Figure 2).
Subsequently, the patient underwent cystourethroscopy
which showed a normal urinary bladder with moderate
hydroureteronephrosis is caused by obstructive retroperitoneal
lymphocele. A double-J stent was then placed to alleviate obstruction.
Follow-up CT guided aspiration of the retroperitoneal collection
yielded 35 mL of clear yellow fluid, which was negative for infection
on culture and sensitivity tests. A CT scans of the abdomen one and
half months later showed a persistent/recurrent retroperitoneal fluid
collection (Figure 3). This, the decision was made to carry out CTguided
percutaneous left retroperitoneal lymphocele aspiration and
drainage catheter placement. Twenty-five mL of clear yellow fluid was
aspirated, an 8 French pigtail drainage catheter was placed (Figure 4),
and sclerotherapy performed using 10 mL of 98% (absolute) ethanol
which was left to dwell for one hour prior to drainage. However,
the lymphocele persisted after five sessions of sclerotherapy with
absolute alcohol. Consequently, the patient underwent left inguinal
lymphangiography and glue embolization of the left retroperitoneal/
iliac supplying lymphatic vessels to treat lymphocele.
Figure 1
Figure 1
Color Doppler ultrasound using a 5 MHz curved transducer shows
left renal hydronephrosis appreciated by renal pelvic dilatation.
Figure 2
Figure 2
A. Transverse view of contrast CT scan show shydronephrosis
and dilatation of the collecting system to the level of the proximal ureter. B.
Coronal view of contrast CT scan demonstrates a 3.6 cm x 4.1 cm x 5.6 cm
hypodense retroperitoneal collection. C. On the transverse view of contrast
CT scan, the left ureter is not distinguishable from fluid collection and the left
ureter is not visualized distal to this cystic fluid collection.
Technique
First, the hilum of a left groin lymph node was accessed with a 9
cm 25-gauge spinal needle under ultrasound guidance (Figure 5A).
Lipiodol was then slowly infused and observed to pass into the left
inguinal lymphatic chains (Figure 5B). After about 30 mins, two
iliac lymphatic vessels were progressively opacified over the mid
left retroperitoneal space to the level of retroperitoneal lymphocele
(Figure 5C). Upon injection of contrast into the left retroperitoneal
cavity, the left lateral retroperitoneal lymphatic vessel was identified
as a potential supplying vessel to the lymphocele (Figure 5D).
The left lateral retroperitoneal/iliac lymphatic vessel, at the level
of L4-L5 and distal to the lymphocele, was successfully accessed
via a 22-gauge Chiba needle through the abdomen under direct
fluoroscopic visualization (Figure 6A). Digital lymphangiogram
demonstrated contrast extravasation into the left retroperitoneal
lymphocele. Next, embolization was performed with 1 ml of N-butyl-
2-cyanoacrylate glue (n-BCA) glue mixed in a 1:1 ratio with lipiodol
along with tantalum powder for improved visualization (Figure 6B).
The glue was seen to distribute evenly within the left retroperitoneal
lateral lymphatic vessel distal to the lymphocele. Contents of the
lymphocele were then aspirated through the left upper back drainage
catheter, confirming the collapse of the left retro peritoneum
lymphocele. Opacified glue was demonstrated to reach the inferior
aspect of the cavity. Follow-up drainage catheter check five days later
showed no further output. The catheter was thus removed.
Figure 3A and 3B
Figure 3A and 3B
A new left ureteral stent is seen in place with no residual
hydronephrosis. The both coronal and transverse views demonstrate
persistent left retroperitoneal fluid collection and a new left ureteral stent,
revealing independent nature of the cystic fluid collection from the left
ureter. These findings also explain left proximal hydroureteronephrosis as a
consequence of mass effect from this fluid collection.
Figure 4A and 4B
Figure 4A and 4B
These transverse views show CT-guided percutaneous
left retroperitoneal lymphocele aspiration and drainage catheter placement
through the left psoas muscle.
Figure 5
Figure 5
A. A left groin lymph node was accessed with a 9 cm 25-gauge
spinal needle under ultrasound guidance. B. Fluoroscopy image showing
intranodal lipiodol injection. C. Extravasation of lipiodol into the soft tissues
is shown. This was corrected by puncturing an adjacent left inguinal lymph
node. Lipiodol seen passing into the left iliac lymphatic vessels. After about
30 minutes, two retroperitoneal/iliac lymphatic vessels are progressively
opacified over the mid left retroperitoneal space, caudal to the retroperitoneal
lymphocele. D. Contrast injection into the cavity via indwelling catheter
identifies left lateral lymphatic vessel as a contributor to the persistent/
recurrent left retroperitoneal lymphocele.
Figure 6
Figure 6
A. Anteroposterior and lateral views. A 22-gauge Chiba needle
was transabdominally advanced with straight approach toward the visualized
mid left retroperitoneal/iliac lymphatic vessel just caudal to the lymphocele.
Digital lymphangiogram performed through the needle shows a single
retroperitoneal/iliac lymphatic vessel, with contrast extravasation into the left
retroperitoneal lymphocele. B. Embolization with 1 ml of n-BCA glue in a 1:1
ratio with lipiodol as well as tantalum powder for improved visualization. Glue
distributing evenly within the left lateral retroperitoneal/iliac lymphatic vessel
caudal to the lymphocele.
Discussion
There are well described techniques in the surgical and
interventional literature regarding treatment of refractory
lymphoceles, the most common of which are surgical resection,
aspiration/drainage, and sclerotherapy [1-5]. However, these
interventions have been associated not only with added risk to the
patient but high treatment failure rates [1-6]. We present a case
of post-surgical retroperitoneal lymphocele that was successfully
treated by lymphatic vessel mapping and glue embolization after five
unsuccessful sessions of sclerotherapy. Sclerotherapy and drainage
are commonly utilized as first line treatment. Chemical sclerotherapy
is usually performed by first aspirating the lymphocele cavity with
subsequent infusion of sclerosant, often requiring repeated sessions.
Commonly used sclerosants include alcohol, povidone iodine,
and doxycycline [12]. Success of sclerotherapy has been found to
be directly related to the size of the lymphocele cavity, with larger
cavities more likely to be resistant to treatment. Complications of
sclerotherapy include catheter related infection, potential allergy or
adverse reaction to sclerosant, and spillage of sclerosant into the retro
peritoneum causing massive inflammation [13]. Glue embolization
on the other hand is an outpatient procedure requiring only one
visit, which eliminates the potential complications from various
sclerosants [14].
With larger lymphoceles or failed sclerotherapy and drainage,
traditional surgical options include laparoscopic surgery, open
marsupialization, and percutaneous imaging guided lymphatic
ligation. These surgical interventions, while typically successful, are
associated with longer hospital stay and increased infection rates.
Additionally, patients must be able to tolerate further surgery,
making this option unavailable for more complicated patients
[15]. Conversely, glue embolization is a two-step procedure during
which one identifies the leaking lymphatic vessel and proceeds with
embolization all in a single session [16].
This renders lymphatic mapping by ultrasound guided intranodal
lymphangiogram and glue embolization a viable alternative to
the established approaches, minimizing the risk of potential
complications and decreasing the length of treatment commonly
associated with surgery and sclerotherapy [17]. Access to the
retroperitoneal lymphocele can be performed with cone beam CT,
temporary catheter placement and subsequent lymphangiogram and
glue embolization with fluoroscopic imaging as a single procedure.
Current noninvasive methods aim treatment at collapsing the walls
of the lymphocele, while a few treatment options target the feeding
lymphatic pathway directly. This is where a minimally invasive
technique such as lymphography and selective glue embolization
of feeding lymphatics may be invaluable as a stand-alone treatment
option, or in conjunction with drainage and sclerotherapy [18], as
was utilized in our case.
This method of selective glue embolization of lymphatic leaks
is well described with thoracic duct lymphangiography for postsurgical
leaks [19,20]. To our knowledge, there is sparse evidence of
the application of this technique elsewhere. In the case of our patient,
a similar technique was applied to the inguinal lymphatic pathway
involving the retro peritoneum. Although lymphangiography and
embolization of the supplying lymph vessels was performed after
sclerotherapy failure, it has the potential to be the first-line treatment.
Main advantage of this technique when compared to sclerotherapy
that it requires only one visit. Minimally invasive nature of the
procedure renders it a very attractive option in comparison to surgery
or sclerotherapy.
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