With the advances in the understanding of anatomy and physiology of the lymphatic system and the pathophysiology of lymphedema newer surgical modalities are being explored. There has been a paradigm shift in the treatment of lymphedema from radical procedures to physiological procedures over the last few decades. Vascularized lymph node transfer (VLNT) is a surgical procedure wherein functional LN from the nonaffected sites are transferred, with micro anastomosis with vessels in the recipient area to physiologically restore the lymphatic flow in an extremity affected by lymphedema. In this case report, we are presenting a case of lower-limb lymphedema which was treated by heterotopic VLNT from the right supraclavicular region based on transverse cervical artery (TCA) and external jugular vein (EJV) to the leg where the vessels were anastomosed with anterior tibial artery and its vena commitante.
The patient was a 17-year-old girl presented with bilateral (right > left) lower-limb edema for 3 years. There was no previous history of trauma or surgery. The patient comes from a region which is endemic for Filariasis in Uttarakhand, India. The edema first started in the foot first in the right leg 2 years ago which gradually ascended to involve her leg and thigh over the last 2 years. Six months after the onset of swelling in right limb, she also started having swelling in the left foot which gradually progressed to involve her left leg up to the knee joint. There is no history of pain or fever. The patient first came to our institute, 6 months ago where she received one course of diethylcarbamazine and was also managed by conservative measures such as limb elevation, therapeutic massage, and compression stockings for 6 months even after which there was no signs of improvement and then she was referred to the plastic surgery department. On examination, the left leg had stage 2, nonpitting, and nonreducible lymphedema after limb elevation. The right leg also had stage 3 lymphedema with same characteristics. Magnetic resonance imaging showed increased subcutaneous tissue with normal underlying muscle. Lymphoscintigram was abnormal and demonstrated impaired lymphatic flow and dermal backflow after radiotracer injection. The patient underwent first author's modification of combined debulking and physiological procedure in the right leg and heterotopic vascular supraclavicular LN transfer in the left leg. Doppler done every day postoperatively until the day of discharge in postoperative day 7 [Video 1] demonstrated the patency of the anastomosis. Postoperatively, the limb was kept in an elevated position and there was significant decrease in the edema in the foot and the nonpitting edema had converted to pitting type of edema. The patient was discharged on postoperative day 7, and suture removal was done on postoperative day 10 both at the donor and recipient site. There was no edema in the donor site, and the scar was well hidden by clothing. On follow-up, there was obvious change in the girth at 9 months postoperatively.
The patient underwent the first author's modification of combined debulking and physiological procedure in the right leg which included the excision of lymphatic tissue, fascia, intermuscular septum, and burying the dermal flap in the intermuscular space created after excision of the septum.
Procedure of vascularized lymph node transfer without skin paddle
A curvilinear incision was marked over the anterior compartments of the left leg after painting and draping, incision was made after infiltration was done with lignocaine 2% with adrenaline solution. Dissection was carried out in the anterior compartment. Extensor digitorum longus muscle was retracted laterally and anterior tibial artery was located. Anterior tibia artery and its venae commitante in midleg prepared as recipient vessels. Curvilinear incision was marked in the posterior triangle in the neck in right side [Figure 1a]. After infiltration with lignocaine 2% with adrenaline subplatysmal flaps were raised. Supraclavicular LN was located. Inferior belly of omohyoid was retracted, and TCA was located arising from the right thyrocervical trunk. Free right supraclavicular LN based on transverse cervical vessels and EJV harvested [Figure 1b]. A curvilinear incision over the anterior compartment of middle third of left leg was made [Figure 2a]. The anterior tibial artery and its venae comittantes were skeletonized. The TCA was anastomosed with the ATA, and the caliber of both these was similar increasing the ease of anastomosis and better chances of flap survival. Venous anastomosis was done between EJV and venae commitante of the ATA. Lateral skin flaps were closed over the transferred LN. Postoperatively, the limb was kept in an elevated position, and compression dressing was avoided to prevent compression at the anastomotic site. Venae commitante and anterior tibial artery anastomosed with EJV and TCA, respectively [Figure 2b]. Good pulsations in the anastomosed vessels and surface oozing from LN containing tissue observed. Skin flaps closed primarily over the transferred LN. The skin flaps in the donor site closed primarily and the resultant scar was barely discernible o follow-up.
Postoperatively, the limb was kept in an elevated position, and no compression dressing was given. Compression dressing was avoided to prevent compression at the anastomosis site. There was a significant decrease in leg circumference and edema over the period of 9 months [Figure 3a-c].
Surgery for lymphedema has tremendously advanced over the past few decades. Surgeons have gradually switched from debulking radical procedures to functional physiologic procedures. Lymphvenous bypass is performed in early cases of lymphedema but is noticed to be less effective in late cases as the lymphatics become sclerosed. In such cases, VLNT is a better option in that it restores the physiologic lymphatic flow. It also has been noticed that these procedures substantially reduce the incidence of frequent episodes of cellulitis through local immunomodulation. This was an ideal case for VLNT because there was significant segmental dermal backflow with very few functional lymphatic vessels on imaging by lymphoscintigraphy. There are various options for donor sites for VLNT s such as the groin, supraclavicular region, axilla, submental region, lateral thoracic region, jejunal mesentery, and greater omentum. Out of these, the LN in the supraclavicular region is preferable due to many reasons. These LN belong to level Vb of the neck nodes and they drain the lung, thyroid, and esophagus, but their removal is barely of any consequence as it is observed that they are routinely removed in oncological cases where neck dissections are done and their removal does not cause significant lymphedema in the neck region. Furthermore, the resultant scar at the donor site is barely discernible and can be concealed easily by the clothing. Unlike groin and axillary regions, there is minimal secondary iatrogenic lymphedema in supraclavicular LN transfer.
Supraclavicular flap based on TCA was first described by Converse and Kazamjian in 1949. Pallua et al. refined this flap, and recently, this flap is being used for vascularized LN transfer. However, we used this flap without skin paddle and monitored through the lateral thinned-out flaps of leg used for tension-free closure over the transferred LN.
There are wide variations in the anatomy of the TCA reported in literature. About 80% of the TCAs arise from thyrocervical trunk, and in remaining 20% cases, they are known to arise from the subclavian artery. In our case, the TCA was seen to arise from the right thyrocervical trunk.
On efforts of search on PubMed, Medline, and Google Scholar, we could find only two cases of LN transfers reported from India, and the authors have transferred the LN from the axillary region in both the cases.
In our case, we found a significant decrease in leg circumference over the period of 9 months [Figure 3a-c]. Furthermore, the lymphedema had decreased from Campisi et al. Stage 2 to Stage 1.
VLNT from the supraclavicular region is a very effective treatment for physiological restoration of the lymphatic flow in established cases of lower-limb lymphedema because of well-hidden favorable scar and minimal chances of secondary iatrogenic lymphedema in the donor region unlike other sites such as the groin and axilla. However, the LN transfer without skin paddle is possible if tension-free closure of soft tissue is done and hence no compression of vascular flow. The VLNT should be preferred over manual lymphatic drainage because it is very effective treatment for physiological restoration of the lymphatic flow, however, the further analytical studies are required to confirm statistically the effectiveness of the same over other modalities.
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Conflicts of interest
There are no conflicts of interest.
Level of evidence: V.
A written informed consent for surgery, clinical photography and publication for academic purposes taken from the patient.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published, and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.
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