Successful Management of a High-output Lymphorrhea via Lymphaticovenous Anastomosis after Cannulation for Cardiopulmonary Bypass

Summary:

Lymphatic leaks are a rare phenomenon, but can be a troublesome and persistent problem, especially in an already debilitated patient. Historically, management of lymphorrhea has involved non- and minimally-invasive techniques of elevation, compression, aspiration, or drain placement, among others. Ligation and sclerotherapy are additional utilized techniques, directly targeting the lymphatic vessel. Microsurgical management of lymphatic leaks via lymphaticolymphatic and lymphaticovenous anastomosis has gained popularity amongst surgeons as an alternative solution to the problem. We present a patient who developed a high-output lymphocutaneous fistula after a femoral cannulation procedure for cardiopulmonary bypass for an orthotopic heart transplantation. After multiple unsuccessful attempts at traditional management options, the patient had a successful resolution of the high-output lymphorrhea via a lymphaticovenous anastomosis utilizing end-to-end coaptation with an interpositional vein graft. This case uniquely describes a lymphaticovenous anastomosis and bypass of a lymph node in the setting of significant lymphorrhea (>1.0 L per day) and associated lymphocutaneous fistula, that was effectively managed in the acute postoperative setting. Management of lymphorrhea by microsurgical techniques and lymphatic vessel manipulation in the postoperative period provides surgeons with an enhanced option for direct operative management of lymphatic vessels and their associated sequelae.

The incidence of groin lymphatic leaks after cannulation procedures for cardiopulmonary bypass in orthotopic heart transplantation is unknown, as there are no published studies looking at this specialized subset of patients. The incidence is likely similar to groin lymphatic leaks after arterial reconstruction in vascular patients, which occurs in 1.8% to 5% of cases.^1,2 Described management options include noninvasive measures such as bedrest, elevation and pressure dressings, whereas more invasive procedures include sclerotherapy, aspiration, drain placement, and vessel ligation surgery.^1–3 Availability and advancements of intraoperative imaging and microscope technologies have provided microsurgeons more opportunities to intervene and directly target the repair of lymphatics in cases of lymphorrhea and chronic lymphedema.^4,5 This case presents intervention of a high-output groin lymphorrhea and associated lymphocutaneous fistula after multiple unsuccessful attempts with noninvasive therapies and attempt at lymphatic vessel ligation. We describe a lymphaticovenous anastomosis, utilizing an interposition vein graft directly establishing a functional lymphatic outflow tract, as a surgical option for the management of acute high-flow lymphorrhea and resolution of an associated lymphocutaneous fistula.

CASE PRESENTATION

The patient is a 52-year-old man with a medical history significant for transposition of great vessels, Eisenmenger syndrome, and pulmonary hypertension, who underwent orthotopic bilateral lung and heart transplantations requiring cardiopulmonary bypass with cannulation via femoral arterial access. The patient’s postoperative course was complicated by multisystem organ failure, sternal dehiscence, tracheal dehiscence, and high-output drainage of both groins, right worse than left. Of note, the patient’s right groin was cannulated using a cut down technique, but the left was cannulated using a Seldinger technique. The patient was found to have dehiscence of his right groin incision with greater than 1.0 L of daily output from the wound. The patient underwent open exploration by the primary team and attempted ligation, without success, followed by lymphangiogram-guided suture ligation by the interventional radiology team with nominal effect. The plastic and reconstructive surgery team was consulted for continued outputs of greater than 1.0 L daily through the right groin 6 weeks after the patient’s index procedure. Due to the length of time between index procedure and consultation, primary anastomotic repair was not feasible due to tissue and vessel retraction in the area.

The patient was taken to the operating room, placed supine, induced under general anesthesia, and a lymphogram using indocyanine green and isosulfan blue was completed. A near-infrared camera (Hamamatsu PDE, Mitaka USA, Denver, Colo.) was then used to identify lymphatic vessels and areas of lymph fluid collection within the right groin. The right groin was explored under the operative microscope (Zeiss Pentero with Foldable Tube and Magnification Exchanger). The donor site morbidity of the vein graft was minimal and simultaneously harvested while the lymphatic and venous recipient sites were being prepared. The area of interest was dissected and four lymphatic vessels containing isosulfan blue were identified converging into what appeared to be a scarred lymph node that was draining copious lymphatic fluid, which was additionally visualized on intraoperative lymphangiography (Fig. 1). Within the wound bed, a vein was identified that did not have the length to reach the lymphatics; therefore a 1.0 mm × 30 mm vein graft was harvested from the forearm. An end-to-end anastomosis was performed using 11-0 nylon suture between the vein graft from the recipient vein. The convergence of lymphatics were then intussuscepted into the vein graft and anastomosed using 11-0 nylon sutures (Fig. 2). The lymphaticovenous anastomosis filled with fluid distal to each anastomosis after patency check effectively bypassing the scarred lymph node. There was no observed leakage of lymphatic fluid following anastomosis on repeat intraoperative lymphangiography. The wound was closed in three layers, and an incisional negative pressure wound therapy dressing was placed.

Fig. 1.

One of the four lymphatic channels, with a clip preventing lymphatic leakage (blue arrow) converging into scarred lymph node (black arrow). An interposition vein graft already sewn into the recipient vein.

Fig. 2.

Completed lymphaticovenous anastomosis with bypass of scarred lymph node and interposition vein graft (black arrow). Lymphatics being intussuscepted (blue arrow).

The patient’s closed incision negative pressure wound therapy dressing was removed on postoperative day 14. The patient had a total of 150 mL of drainage over the 2-week period the negative pressure wound therapy was in place, with the majority of the drainage during the first 2 postoperative days The patient’s prealbumin rebounded two points in the first week after surgery (Fig. 3). Three weeks from surgery, a groin collection re-accumulated, and an interventional radiology drain was placed into the right groin and underwent three trials of doxycycline sclerotherapy without any changes to the patient’s prealbumin, which led us to believe our anastomosis was still intact. However, due to persistent groin fluid collection, it was decided that we would take the patient back to the operating room for an exploration. Upon evaluation in the operating room there was noted to be a residual Dacron patch visible in the wound bed. Vascular surgery was consulted intraoperatively for removal of the patch. After patch removal, there was no persistent fluid accumulation, and the wound bed was clean and dry; the anastomosis was therefore not manipulated so as to preserve its healing. The area was then closed with a rotational rectus femoris muscle flap for minimization of dead space. Since the second surgery and removal of the Dacron patch, the patient has been doing well. The patient was seen 10 months postoperatively and had maintained resolution of his groin incision without evident of lymphedema.

Fig. 3.

Prealbumin trend during hospital course with dates labeled on the x-axis. The red line represents the first operation (lymphaticovenous anastomosis and lymph node bypass), and the green line represents the second operation (Dacron graft removal and rectus femoris muscle flap).

DISCUSSION

Lymphaticolymphatic and lymphaticovenous anastomotic surgery was originally described in the 1970s to treat obstructive lymphedema.⁶ With advances and increased popularity in microsurgery, this technique has been demonstrated to be helpful and successful in the treatment of lymphedema and lymphorrhea patients.^3,4,7 Given the advances in lymphatic surgery, new applications of lymphatic surgery will continue to arise and may be helpful in situations similar to this case report.^5,7,8 As groin lymphatic lymphoceles or lymphocutaneous fistulas can be quite cumbersome in critically ill patients; treatment occurs in a stepwise fashion with conservative measures being attempted first to prevent the patient from undergoing more invasive measures.² When surgery is considered, it can be as minimal as an incision and drainage procedure or ligation, versus a larger surgery that may require a flap. Local muscle flaps such as sartorius, gracilis, rectus abdominis, or rectus femoris have been described for such uses.^9,10 In our patient, we successfully treated a high-output lymphorrhea and associated lymphocutaneous fistula with lymphaticovenous anastomosis, utilizing a vein interposition graft. Additionally, we used a rotational rectus femoris muscle flap which reduced dead space and supported the area, providing complimentary support to the anastomosis. This case provides dramatic resolution of, to our knowledge, the highest output lymphorrhea described along with a bypass of a nonfunctional node.

CONCLUSION

This technique can provide a powerful option for surgeons managing complex lymphorrhea, lymphocele, or lymphocutaneous fistulae, and we advocate for its consideration as part of the treatment algorithm.