Lymphovenous Bypass Using Indocyanine Green Mapping for Successful Treatment of Penile and Scrotal Lymphedema

Abstract

Male genital lymphedema is a debilitating condition with significant physiologic and psychologic ramifications. Classical surgical treatments for male genital lymphedema include primarily ablative procedures through removal of excess soft tissue, which often have poor aesthetic and functional outcomes. Super microsurgical techniques (including lymphovenous bypass and lymph node transfers) are promising contemporary interventions. In this case report, we aim to share our experience of lymphovenous bypass with indocyanine green (ICG) lymphangiography in the management of penile and scrotal lymphedema. We performed ICG lymphography of the male genitalia and right thigh by injecting ICG at multiple sites followed by concomitant evaluation with a handheld fluorescent portable imager. Skin incisions were designed over the linear lymphatics upstream from the site of obstruction and dermal backflow. Four end-to-end and one end-to-side lymphovenous bypasses were performed. After completion, lymphovenous bypasses patency was confirmed by injecting ICG proximal to the incision and observing flow. At 10-month clinic follow-up, the patient showed marked improvement with improved skin tenting, softer tissues, improved sensation, visible dorsal penile vein, ability to retract foreskin for cleaning, and confidence to engage in sexual activities. This case report describes successful use of lymphovenous bypass in the treatment of penile and scrotal lymphedema using ICG lymphography intraoperatively to map functioning of superficial lymphatics. The full potential of this microsurgical approach is yet to be discovered, and future studies are needed to enhance the long-term outcomes for the treatment of penoscrotal lymphedema.

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Reconstructive
From the Division of Plastic Surgery, Saint Louis University School
of Medicine, St. Louis, Mo.
Received for publication April 6, 2020; accepted May 5, 2020.
This work was conducted in accordance with ethical standards.
Copyright © 2020 The Authors. Published by Wolters Kluwer Health,
Inc. on behalf of The American Society of Plastic Surgeons. This
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DOI: 10.1097/GOX.0000000000002938
INTRODUCTION
Male genital lymphedema is a debilitating condition
with signicant physiologic and psychologic ramica-
tions. Classical surgical treatments for male genital lymph-
edema include primarily “ablative” procedures through
removal of excess soft tissue, which often have poor aes-
thetic and functional outcomes.1,2 Lymphovenous bypass
and vascularized lymph node transplants are microsur-
gical techniques typically used to manage lymphedema
of the extremities. Lymphovenous bypass includes the
anastomoses of lymphatic vessels to adjacent subdermal
venules to permit or “bypass” lymphatic drainage into
the venous circulation. Originally described in 1985, few
reports have been published on lymphovenous bypass
for the treatment of primary or secondary male genital
lymphedema.3 Aulia4 performed a recent review of surgi-
cal management for male genital lymphedema and found
that while both ablative and physiologic surgeries pro-
vided some symptom relief, lymphovenous bypass cases
had no recurrence of symptoms at 1-year follow up, com-
pared with rates up to 25% in the ablative group. To our
knowledge, few reports have described the utilization of
indocyanine green (ICG) lymphangiography for intraop-
erative planning in male genital lymphedema surgery.2
ICG uorescence lymphangiography can be used to pro-
vide real-time, high-resolution intraoperative visualization
of the functioning lymphatic system while avoiding expo-
sure to ionizing radiation.3 Further, ICG lymphangiogra-
phy can provide a snapshot of nonfunctional lymphatics
Gianfranco Frojo, MD
Oscar Castro, BS
Kashyap Komarraju Tadisina, MD
Kyle Y. Xu, MD
Summary: Male genital lymphedema is a debilitating condition with signicant
physiologic and psychologic ramications. Classical surgical treatments for male
genital lymphedema include primarily ablative procedures through removal of
excess soft tissue, which often have poor aesthetic and functional outcomes. Super
microsurgical techniques (including lymphovenous bypass and lymph node trans-
fers) are promising contemporary interventions. In this case report, we aim to
share our experience of lymphovenous bypass with indocyanine green (ICG) lym-
phangiography in the management of penile and scrotal lymphedema. We per-
formed ICG lymphography of the male genitalia and right thigh by injecting ICG
at multiple sites followed by concomitant evaluation with a handheld uorescent
portable imager. Skin incisions were designed over the linear lymphatics upstream
from the site of obstruction and dermal backow. Four end-to-end and one end-
to-side lymphovenous bypasses were performed. After completion, lymphovenous
bypasses patency was conrmed by injecting ICG proximal to the incision and
observing ow. At 10-month clinic follow-up, the patient showed marked improve-
ment with improved skin tenting, softer tissues, improved sensation, visible dorsal
penile vein, ability to retract foreskin for cleaning, and condence to engage in
sexual activities. This case report describes successful use of lymphovenous bypass
in the treatment of penile and scrotal lymphedema using ICG lymphography intra-
operatively to map functioning of supercial lymphatics. The full potential of this
microsurgical approach is yet to be discovered, and future studies are needed to
enhance the long-term outcomes for the treatment of penoscrotal lymphedema.
(Plast Reconstr Surg Glob Open 2020;8:e2938; doi: 10.1097/GOX.0000000000002938;
Published online 24 July 2020.)
Lymphovenous Bypass Using Indocyanine Green
Mapping for Successful Treatment of Penile and
Scrotal Lymphedema
CASE REPORT

PRS Global Open • 2020
2
and facilitate the localization of potential target areas for
lymphovenous bypass. In this case report, we aim to share
our experience of lymphovenous bypass with ICG lym-
phangiography in the management of penile and scrotal
lymphedema.
CASE PRESENTATION
A 27-year-old man presented with a 2-year history of
penile and scrotal lymphedema. He reported signicant
pain and discomfort performing manual work, locore-
gional numbness, poor foreskin hygiene, difculty wearing
clothes, and sexual dysfunction. The patient had previ-
ously been evaluated by 2 urologists and infectious disease
specialists. Evaluations including computed tomography,
hemogram, complete blood count, urinalysis, urine cul-
ture, and electrocardiogram were normal. Interventions
included a 2-week course of furosemide without improve-
ment of symptoms. The patient was further evaluated and
treated by a lymphedema therapist without signicant
improvement. The lymphedema continued to worsen
with eventual progression and involvement of the proxi-
mal right thigh.
Preoperatively, the patient underwent lymphoscintigra-
phy with injection of Tc-99m tilmanocept, which revealed
obstruction with compensatory collateral ow of bilateral
inguinal regions. The patient was scheduled for intra-
operative lymphangiography and lymphovenous bypass.
Intraoperatively, ICG lymphography was performed on
multiple injection sites, including the right hemiscrotum,
ventral penis, dorsal penis, right foot rst web space, right
medial ankle, and right medial thigh. The 6 injection sites
were administered with 0.1 mL of ICG at each respective
location, and uorescence lymphography was performed
using a handheld portable imager (SPY Portable Handheld
Imager, Styker, Kalamazoo, Mich.). Figure1 demonstrates
the mapped lymphatic ow in red and planned incision
markings in blue. Linear ow from the genital injection
sites was followed to the right groin, where abnormal ret-
rograde ow into the dermis and right thigh was noted.
Signicant dermal backow into the dermis of the penis
and scrotum was also noted. Lymphography of the right
leg was performed by injection of the foot rst web space,
which revealed linear lymphatics until marked dermal
backow was again noted in the proximal right thigh.
Skin incisions were designed over the linear lymphatics
upstream from the site of obstruction and dermal back-
ow. After injection of 1% lidocaine with 1:100,000 epi-
nephrine, isosulfan blue was injected upstream to each
incision to identify the lymphatics. Four end-to-end and
one end-to-side lymphovenous bypasses were performed.
The end-to-side anastomosis was performed at the base of
the right hemiscrotum. After completion, patency with-
out leak was conrmed by injecting ICG proximal to the
incision and observing ow using the ICG camera on the
microscope. The patient was discharged home the same
day, with instructions to continue aspirin 325 mg for 30
days, maintain penoscrotal elevation, and follow-up in
clinic. At 10-month clinic follow-up, the patient showed
marked improvement with improved skin tenting, softer
tissues, improved sensation, visible dorsal penile vein,
ability to retract foreskin for cleaning, and condence
to engage in sexual activity. Figure2 demonstrates a side-
by-side comparison of patient photographs taken on the
day of surgery and taken after 10-month follow-up. The
patient was able to return to all normal activities and was
pleased with the results of surgery.
DISCUSSION
As initially described by Sappey and Mascagni, the
supercial and deep lymphatic pathways provide 2
options for lymphovenous shunting in the male genital
region.1 In a series of 3 patients, Otsuki et al supported
utilization of the higher caliber, deep lymphatic system
as opposed to the potentially degenerated supercial
lymphatic system as described by Mukenge and Huang
in separate years prior.1,3,5 However, deep inguinal lym-
phatics are close in proximity to the femoral vessels,
potentially increasing the risk of iatrogenic injury dur-
ing exploration. With utilization of ICG lymphography,
we identied and used the functioning supercial lym-
phatic vessels in a single male patient with secondary
penoscrotal lymphedema similar to lymphography eval-
uation previously described by Yamamoto et al.6 Due to
the visible supercial linear lymphatics, we elected to
proceed with multiple lymphovenous bypasses for the
treatment of penoscrotal lymphedema as previously
described in the literature.7
Fig. 1. Intraoperative ICG lymphatic mapping and incision planning.
A, ICG lymphography demonstrates linear lymphatics and areas of
dermal backow. B, Red marking indicates linear lymphatics, and
blue marking indicates nal planned incisions.

Frojo et al. • Lymphovenous Bypass Penile and Scrotal Lymphedema
3
Guiotto et al8 performed a systematic review of out-
comes after genital lymphedema surgery, which exam-
ined 20 published reports, including a total of 151
patients. Three main surgical treatment groups were
identied, including resection and primary closure or
skin graft, resection followed by ap reconstruction, and
lymphovenous bypass. Lymphovenous bypass was found
to have the lowest complication rate (9%), but it was
only performed in 14.5% of the total cases. The study
highlighted the lack of consensus recommendations
for preoperative assessment and surgical management
of patients with genital lymphedema.8 Based on our
limited experience in genital lymphedema surgery, we
advocate that ICG lymphography is a reliable method to
characterize both normal and abnormal lymphatic ow,
as well as provide microsurgeon’s assistance in selecting
sites for lymphovenous bypass within the supercial lym-
phatic system.
CONCLUSIONS
This case report describes successful use of lympho-
venous bypass in the treatment of penile and scrotal
lymphedema using ICG lymphography intraoperatively
to map functioning supercial lymphatics. The full
potential of this microsurgical approach is yet to be dis-
covered, and future studies are needed to enhance the
long-term outcomes for the treatment of penoscrotal
lymphedema.
Kyle Y. Xu, MD
Division of Plastic Surgery
Department of Surgery
Saint Louis University School of Medicine
3635 Vista Avenue
St. Louis, MO 63104
E-mail: kyle.xu@health.slu.edu
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Fig. 2. Preoperative and postoperative comparison of the patient’s genitalia. A, Anterior view of penis
and scrotum on the day of surgery. B, Anterior view of penis and scrotum on postoperative 10-month
clinical follow-up.