Abstract
Objective:
To assess lymphangiography findings and outcome of lymphatic embolisation to manage chyle leak after neck surgery.
Methods:
Consecutive cases of lymphangiography performed between April 2018 and May 2022 for management of chyle leaks related to neck surgery were retrospectively reviewed. Lymphangiography findings, techniques, and outcomes were analysed.
Results:
Eight patients (mean age: 46.5 years) were included. Six patients had undergone radical neck dissection for thyroid cancer, and two had undergone lymph node excision. Clinical presentations were: chyle drainage through Jackson Pratt catheters in five patients, lymphorrhea through surgical wounds in two, and enlarging lymphocele in one. Lymphangiography techniques included: inguinal lymphangiography in four patients, retrograde lymphangiography in three, and transcervical lymphangiography in one. Lymphangiography revealed leaks in the terminal thoracic duct in two patients, bronchomediastinal trunk in two, jugular trunk in three, and superficial neck channels in one. Embolisation techniques included: non-selective embolisation of terminal thoracic duct (n = 2), selective embolisation of the jugular trunk (n = 3), selective embolisation of the bronchomediastinal trunk (n = 2), and intranodal glue embolisation of superficial neck channels (n = 1). One patient underwent a repeat procedure. Chyle leak resolved in all patients over a mean of 4.6 days. No complication was encountered.
Conclusion:
Lymphatic embolisation seems to be effective and safe in managing chyle leaks after neck surgery. Lymphangiography allowed for the categorisation of chyle leaks according to their location. Post-embolisation patency of the thoracic duct may be preserved in chyle leaks that do not directly involve the thoracic duct.
Advances in knowledge:
Lymphatic embolisation is safe and effective in managing chyle leaks after neck surgery. On lymphangiography, the location of contrast media extravasation may not be consistent. The technique for embolisation should be based on the location of the leak. Post-embolisation patency of the thoracic duct may be preserved in chyle leaks that do not directly involve the thoracic duct.
Introduction
Chyle leak after neck surgery is a rare complication of thyroidectomy and neck dissection with a reported incidence of 0.5–8%. 1 Typical manifestations are skin lymphorrhea from the surgical wound, chylous drainage through the surgical catheter, and enlarging lymphocele. Long-term complications include failed wound healing, malnutrition, and immunosuppression. 1,2 Conservative management involving diet restrictions has been shown to be effective in low-output leaks. However, it is time-consuming and may potentially fail in high-output leaks. 3 Surgical ligation of the thoracic duct is considered in cases where conservative treatment fails. 4,5 Thoracic duct embolisation has been proposed as an alternative to thoracic duct ligation. 6–10 Despite accumulating data on the outcome of thoracic duct embolisation for chylothorax, literature on its application for chyle leak in the neck is limited. 11–13 Moreover, previous reports do not provide a detailed description of the location of leaks on lymphangiography. In this study, findings on lymphangiography, type of embolisation technique, and treatment outcome were reviewed.
Methods and materials
Patients
A retrospective review of consecutive lymphangiography cases performed between April 2018 and May 2022 revealed eight patients who had been referred for chyle leak after neck surgery. Institutional Review Board approval was obtained, and the requirement for patient consent was waived due to the retrospective nature of this study. Patient demographics are presented in Table 1. The mean age of the group was 46.5 years (range: 22–61 years), and the male-to-female ratio was 1:1. Six patients had undergone radical neck dissection due to thyroid cancer, and two others had undergone excision of cervical lymph nodes revealing metastatic malignancy. Clinical presentations included: chyle drainage through Jackson Pratt catheters in five patients, lymphorrhea through skin wounds in two patients, and enlarging lymphocele in one patient. Indication for lymphangiography was as follows: (1) chyle drainage >1000 ml/24 h, (2) chyle drainage >300 ml/24 h persisting longer than a week despite conservative treatment consisting of a low-fat diet supplemented with triglycerides and intravenous octreotide. For lymphorrhea occurring in the surgical wound, the amount of chyle leak could not be measured because the leak presented as oozing lymph from the surgical wound. Indication for lymphangiography was when there was persistent oozing of chyle from the surgical wound for over a week. Total parenteral nutrition was additionally prescribed when chyle drainage exceeded 1000 ml/24 h.
Table 1.
Baseline demographics and clinical outcome
| Patient no. | Age /Sex | Primary disease | Surgery | Symptoms | BP (PR) | Hb/Hct | PLT | PT/INR | Time between surgery and intervention (days) | Output one day before intervention (mL) |
|---|---|---|---|---|---|---|---|---|---|---|
| 1 | 41 /M | PTC | Total thyroidectomy, bilateral CCND, left mRND | Chyle drainage | 135/85 (65) | 14.4/41.9 | 213 | 11.8/1.10 | 3 | 1156 |
| 2 | 57 /F | Lung cancer | Surgical excision of metastatic cervical LN | Lymphorrhea | 106/60 (89) | 12.7/40.5 | 210 | 11.5/1.00 | 13 | 130 b |
| 3 | 49 /F | PTC | Total thyroidectomy, bilateral CCND | Chyle drainage | 172/84 (68) | 13.3/39.9 | 206 | 12.6/0.99 | 8 | 563 |
| 4 | 40 /M | PTC | Total thyroidectomy, bilateral CCND and mRND, and mediastinal LN dissection | Lymphocele | 108/67 (75) | 12.3/37.5 | 173 | 13.2/1.05 | 85 | N/A a |
| 5 | 42 /M | PTC | Total thyroidectomy, bilateral CCND, and right mRND | Lymphorrhea | 160/73 (75) | 14.9/44.1 | 239 | 12.3/0.96 | 9 | 60 b |
| 6 | 22 /F | PTC | Total thyroidectomy, bilateral CCND, left mRND | Chyle drainage | 129/69 (64) | 12.4/37.8 | 219 | 13.5/1.07 | 10 | 375 |
| 7 | 61 /F | Ovarian cancer | Surgical excision of metastatic cervical LN | Chyle drainage | 119/79 (77) | 11.6/34.7 | 252 | 12.2/0.94 | 15 | 500 |
| 8 | 60 /M | PTC | Total thyroidectomy, bilateral CCND, left mRND | Chyle drainage | 151/74 (73) | 15.5/45.4 | 223 | 12.7/1.00 | 7 | 1030 |
BP, blood pressure (indicated as systolic/diastolic pressures in mmHg); CCND, central cervical node dissection; Hb/Hct, haemoglobin/haematocrit (g/dL); LN, lymph node; N/A, not applicable; PLT, platelet count (x10³/uL); PR, pulse rate (beats per minute); PTC, papillary thyroid cancer; PT/INR, prothrombin time in sec/international normalised ratio); mRND, modified radical neck dissection.
Daily output before day of intervention is not available because the lymphocele was drained at the time of lymphatic intervention (13 ml of chylous fluid was aspirated on site).
Output could not be accurately measured in lymphorrhea due to chyle oozing from the surgical wound. Frequent change of dressing was required due to oozing chyle.
Intranodal lymphangiography
Intranodal lymphangiography was performed either in the groin or neck under real-time ultrasound guidance using a high-frequency (7–15 MHz) linear transducer (HD 11 XE; Philips, Amsterdam, the Netherlands). The tip of a 25-gauge needle was positioned inside the target lymph node after which Lipiodol (Guerbet, Roissy, France) was slowly injected into the lymph node to opacify the lymphatic system. The movement of Lipiodol was observed under intermittent fluoroscopy until the region of interest was opacified and until the leak was detected.
Retrograde lymphangiography
Direct transcervical access into the terminal thoracic duct was achieved by guiding a 21-gauge micropuncture needle (Cook, Bloomington, IN) under real-time ultrasound guidance. A 0.016-inch guide wire (Asahi Meister; Asahi. Intecc Co., Ltd., Nagoya, Japan) was inserted through the needle into the thoracic duct. The guide wire and microcatheter (Progreat; Terumo, Tokyo, Japan) were navigated in a retrograde fashion into the lower thoracic duct.
Transvenous access into the terminal thoracic duct was performed by accessing the right femoral vein and directing a 4-Fr angiographic catheter to the lymphovenous junction under fluoroscopy. After engaging the lymphovenous junction with the angiographic catheter, a coaxial microcatheter and guidewire system was advanced into the terminal thoracic duct. Lymphangiography of the thoracic duct was performed with water-soluble, iodine contrast media using digital subtraction mode on the angiography equipment.
Thoracic duct access
For antegrade thoracic duct access, the cisterna chyli that was opacified through intranodal lymphangiography was punctured through the peritoneum under fluoroscopic guidance using a 21-gauge Chiba needle (Cook, Bloomington, IN). An 0.014-inch (Command; Abbott Vascular, IL) or 0.018-inch (V18; Boston Scientific, Natick, MA) guidewire and microcatheter were then passed into the thoracic duct.
Technical details for retrograde thoracic duct access are described in the previous section. An alternative technique for retrograde access was through the Jackson Pratt drainage catheter. The exposed end of the drainage catheter was punctured to allow the passage of a microcatheter through the drainage catheter lumen. Thereafter, the microcatheter was redirected outside the drainage catheter lumen through one of the distal side-holes and navigated into the leaking duct.
Embolisation
As soon as the location of the leak was identified on lymphangiography, embolisation was performed in the same sitting. The microcatheter was navigated as close to the leak as possible, followed by injection of a glue mixture with or without the placement of fibered coils (Nester; Cook, Bloomington, IN, or Concerto; Medtronic, Minneapolis, MN). The glue mixture was created by diluting N-butyl cyanoacrylate (n-BCA) glue (B. Braun, Melsungen, Germany) with Lipiodol (mixture ratio ranged from 1:1 to 1:4 depending on leak location). The microcatheter was flushed with dextrose-5-water prior to glue injection in order to prevent premature polymerisation of glue mixture inside the microcatheter lumen. In the exceptional case of intranodal glue injection, the lymph node and connecting channels were flushed with dextrose-5-water through the needle, followed by 1:6 glue injection. In all cases of glue embolisation, the glue mixture prepared in a 3 ml syringe was manually injected until the glue cast was demonstrated at the site of the leak.
Assessment of outcome
Chyle drainage was monitored daily before and after the procedure. Medical documents were reviewed to assess the outcome and complications related to the procedure. Clinical success was defined as resolution of chyle drainage leading to successful catheter removal.
Results
Baseline demographics and clinical outcomes are presented in Table 1. Lymphangiography and embolisation were technically successful in all eight patients (100%, 95% CI: 63–100). Intranodal lymphangiography was performed in the groin in four patients and the neck in one patient. Retrograde lymphangiography was performed in three patients: direct transcervical access into the terminal thoracic duct in two patients and transvenous thoracic duct catheterisation in the other. Lymphangiography revealed leaks in the following locations: the main trunk of the terminal thoracic duct in two patients, the bronchomediastinal trunk in two, the jugular trunk in three, and superficial lymphatic channels in the left lateral neck in one. Five different techniques were used for embolisation. In two patients with leaks in the main trunk of the thoracic duct, the terminal thoracic duct was embolised using a combination of coils and 1:1 or 1:2 glue mixture. One of these patients had a lymphocele drained on site (Figure 1) by placing a pigtail drainage catheter. The catheter was successfully removed 4 days after the thoracic duct embolisation. The other patient who underwent thoracic duct embolisation underwent a repeat procedure due to initial failure. The amount of drainage initially decreased 2 days after the first procedure, but it increased on the third post-embolisation day. The chyle leak was monitored for an additional 6 days until it was decided that a repeat procedure was necessary. In the second procedure, direct transcervical access was performed with a micropuncture needle under fluoroscopy-guidance since the residual glue cast from the previous procedure was readily visible under fluoroscopy. A 1:1 glue mixture was subsequently injected through the needle to embolise the terminal thoracic duct. In three patients, the jugular trunk was selectively embolised to treat chyle leak using glue with or without coils (Figure 2). In one of these cases, the jugular trunk was accessed through the Jackson Pratt catheter tract after failed attempts at transperitoneal and retrograde techniques for thoracic duct access (Figure 3). Transperitoneal access was unsuccessful owing to the small size of the cisterna chyli. Transvenous access was unsuccessful because the lymphovenous junction could not be engaged with the angiographic catheter. In two patients with leaks from the bronchomediastinal trunk, the microcatheter was advanced to the confluence of the bronchomediastinal trunk, and 1:4 glue mixture was delivered distally into the bronchomediastinal tributaries (Figure 4). For the last patient, glue embolisation was performed directly through a needle placed in the cervical lymph node (Figure 5). A 1:6 glue mixture was injected until it reached the leak site in the lateral neck. Overall, post-embolisation patency of the terminal thoracic duct was spared in six patients (75%). Technical details are summarised in Table 2. Catheters were successfully removed in all patients within 1–5 days (mean: 2.9 days) after embolisation. No recurrence was reported over a mean follow-up period of 608.6 days (range: 6–1,388 days). No complication (0%, 95% CI: 0–37) was reported during this period.
Figure 1.
A 40-year-old male patient developed chyle leak after total thyroidectomy with radical neck dissection for papillary thyroid carcinoma (patient no. 4). (a) The thoracic duct was directly accessed under ultrasound-guidance, and a microcatheter was navigated into the lower part of the thoracic duct. Digital subtraction lymphangiography was performed. A 7-Fr pigtail drainage catheter (arrow) marks the location of the lymphocele in relation to the terminal thoracic duct (b). Thoracic duct embolisation was performed using coils and 1:1 glue mixture. Extravasated glue cast is noted within the lymphocele (arrow).
Figure 2.
A 61-year-old female patient with underlying ovarian cancer developed chyle leak after surgical excision of a cervical lymph node (patient no. 7). (a) Transvenous, digital subtraction lymphangiography in the terminal thoracic duct (arrowheads) demonstrates extravasation of contrast media from the jugular trunk (arrow). (b) The jugular trunk (arrowhead) was selectively catheterised with a microcatheter. 1:2 glue mixture was slowly injected until the glue mixture reached the leak (arrow).
Figure 3.
A 22-year-old female patient developed chyle leak after total thyroidectomy with radical neck dissection for papillary thyroid carcinoma (patient no. 6). (a) Inguinal lymphangiography reveals extravasation of Lipiodol (arrowhead) from the jugular trunk (arrow). (b) A microcatheter was navigated through the Jackson Pratt catheter (arrowheads) to access the leak. Embolisation was performed using coil and 1:2 glue mixture.
Figure 4.
A 41-year-old male patient developed chyle leak after total thyroidectomy with radical neck dissection for papillary thyroid carcinoma (patient no. 1). (a) Lipiodol lymphangiography via transperitoneal access reveals contrast media extravasation (arrow) from a tributary of the bronchomediastinal trunk. A microcatheter (arrowheads) has been positioned in the bronchomediastinal trunk for embolisation. (b) 1:4 glue mixture was injected into the bronchomediastinal trunk (white arrowheads) until it reached the leak sites (arrows). The thoracic duct (black arrowheads) is demarcated by the Lipiodol column. (c) Cone beam CT demonstrates glue cast in the leak (arrow) and tributary (arrowheads) of the bronchomediastinal trunk.
Figure 5.
A 57-year-old female patient underlying lung cancer developed chyle leak after surgical excision of a cervical lymph node (patient no. 2). (a) A cervical lymph node (arrow) medial to the surgical wound was directly accessed with a 25-gauge needle. Lipiodol lymphangiography demonstrates chyle leak (arrowhead) in the superficial lymphatic channels of the neck. (b) Embolisation was performed by injecting 1:6 glue mixture into the cervical lymph node. Glue mixture was injected until it reached the leak site (arrowhead).
Table 2.
Technical details of lymphangiography and embolisation
| Patient No. | Leak location | Embolisation access | Embolic agent | Embolisation technique | Post-embolisation interval until successful catheter removal (days) |
|---|---|---|---|---|---|
| 1 | Bronchomediastinal trunk | CC | 1:4 glue | Selective embolisation | 2 |
| 2 | Superficial lymphatics in left lateral neck | Cervical LN | 1:6 glue | Intranodal embolisation | 2 |
| 3 | Jugular trunk | CC | Coils and 1:2 glue | Selective embolisation | 1 |
| 4 | Terminal TD leak with lymphocele | TD direct puncture | Coils and 1:1 glue | TDE | 4 |
| 5 | Bronchomediastinal trunk | TD direct puncture | 1:4 glue | Selective embolisation | 4 |
| 6 | Jugular trunk | JP catheter tract | 1:2 glue | Selective embolisation | 3 |
| 7 | Jugular trunk | Transvenous | 1:2 glue | Selective embolisation | 5 |
| 8 | Terminal TD | CC | Coils and 1:2 glue | TDE | N/Aa |
| 8-2 | Terminal TD (recurrent leak) | TD direct puncture | Glue 1:1 | TDE (repeat procedure) | 16b |
CC: cisterna chyli, JP: Jackson Pratt,LN: lymph node, N/A: not application; TD: thoracic duct, TDE: thoracic duct embolisation.
Catheter was not removed due to initial failure. The patient underwent a second procedure nine days after the initial procedure.
Interval between first procedure and catheter removal.
Discussion
Clinical success was achieved in all patients within 1–16 days following embolisation. One of these patients required a repeat procedure. While thoracic duct embolisation is no longer considered a novel procedure, its application to chyle leak related to head and neck surgery has received relatively less attention when compared to its well-established role in chylothorax. 11–14 The reported outcomes of lymphangiography and embolisation for post-operative leaks in the head and neck are summarised in Table 3. 11–16 Since the first reported case of thoracic duct embolisation by Cope et al, 6,7 various techniques for thoracic duct access and embolisation have been developed over the recent years to treat chylothorax and chylous ascites. 17–20 In this study, such techniques were adopted to treat chyle leaks in the neck. In the earlier years of the study, inguinal lymphangiography followed by transperitoneal catheterisation of the thoracic duct was the primary technique. This was recently replaced by retrograde techniques, while the former was reserved for when retrograde access was unsuccessful. Unlike thoracic duct injury resulting from surgical procedures that involve mediastinal structures or lungs, lymphatic injury after neck surgery is likely to occur in the terminal thoracic duct or its tributaries. Retrograde access may be more practical under the circumstances since inguinal lymphangiography and transperitoneal access may be omitted. The feasibility of retrograde access for the management of chylothorax has been demonstrated in previous reports. 17,19
Table 3.
Summary of outcomes in the literature
| Author | Article type | No. of cases | Presentation | Leak location | Catheterisation technique | Embolisation technique | No. of repeat procedures | No. of technically unsuccessful TDE | Clinical success (including LG only) |
|---|---|---|---|---|---|---|---|---|---|
| Patel et al. 15 (2008) | Case report | 1 | JP leak | Terminal TD | Antegrade | Embolisation of main thoracic duct | 0 | 0 | 100% |
| Chen et al. 11 (2015) | Case report | 1 | Neck leak | Terminal TD | Not performed | Not performed | 0 | 0 | 100% |
| Arslan et al. 14 (2017) | Case report | 1 | JP leak | Branch of thoracic duct | Retrograde (transvenous) = 1 | Selective branch embolisation | 0 | 0 | 100% |
| van Goor et al. 16 (2007) | Case series, retrospective | 2 | Neck leak = 2 | Terminal TD | Antegrade | Embolisation of main thoracic duct | 1 | 0 | 100% |
| Moussa et al. 12 (2020) | Case series, retrospective | 6 | Neck leak = 5 Chylothorax = 1 |
Not individually presented | Antegrade | Embolisation of main thoracic duct | 1 | 1 | 100% |
| Ushinsky et al. 13 (2021) | Case series, retrospective | 12 | JP leak = 11 Chylothorax = 1 |
Not individually presented | Antegrade = 8 Retrograde = 1 Both = 1 Unsuccessful = 1 Not performed = 1 |
Embolisation of main thoracic duct = 11 | 3 | 1 | 83% |
LG: lymphangiography, TDE: thoracic duct embolisation, JP: Jackson Pratt
Based on lymphangiography, chyle leaks were categorised according to their anatomical location. Detailed anatomy of the terminal thoracic duct has been described elsewhere. 20–24 Leaks were demonstrated in four locations: the thoracic duct, jugular trunk, tributaries of the bronchomediastinal trunk, and superficial lymphatic channels in the left lateral neck (Figure 6). The embolisation technique was based on the leak’s location. Such a strategy has not been described elsewhere. The main trunk of the thoracic duct was embolised by the conventional technique of thoracic duct embolisation using coils and concentrated glue. For bronchomediastinal trunk leak, the microcatheter was navigated to the insertion point of the bronchomediastinal trunk, and diluted glue mixture was slowly injected into the distal tributaries. Cases with leaks from the jugular trunk were treated by selective embolisation of the jugular trunk without occluding the thoracic duct. Meanwhile, lymphorrhea in the lateral neck was treated by intranodal glue embolisation. This technique was based on that described in the treatment of groin lymphorrhea. 25,26 Overall, post-embolisation patency of the thoracic duct was preserved in all but two patients. This stands out from the technique described in most publications where the thoracic duct was non-selectively embolised to treat chyle leak after neck surgery. A review of the literature disclosed two isolated case reports on selective embolisation of chyle leak where thoracic duct patency was preserved. 14,27 Occlusion of lymphatic outflow to the systemic circulation remains one of the concerns of thoracic duct embolisation, and the true incidence of complications related to this procedure is still debated. 28 Selective embolisation of leaks with preservation of the thoracic duct is appealing because the natural physiology of lymph flow is preserved.
Figure 6.
A schematic representation of the terminal thoracic duct and its tributaries. Lymphangiography revealed leaks in: (a) terminal thoracic duct, (b) bronchomediastinal trunk, (c) jugular trunk, (d) superficial lymphatic channels.
Clinical failure could be avoided with the use of multiple techniques. Any attempt that failed was complemented by the other to overcome technical difficulty. In patient no. 8, retrograde access was unsuccessful owing to transection of the terminal thoracic duct. Thoracic duct embolisation was achieved by inguinal lymphangiography and transperitoneal thoracic duct access. Although the chyle leak initially demonstrated response to thoracic duct embolisation, the leak increased 3 days after the procedure suggesting recanalisation of the thoracic duct. It was presumed that the 1:2 glue mixture that was injected during the procedure was partly washed out from the terminal thoracic duct. As a result, a second procedure was carried out where the glue cast was reinforced by injection of 1:1 glue mixture into the terminal thoracic duct under fluoroscopic guidance. In patient no. 6, antegrade and retrograde access into the thoracic duct were both unsuccessful. Clinical failure was avoided by navigating a microcatheter through the Jackson Pratt catheter tract and into the jugular trunk, where the leak was identified. A reverse approach for glue embolisation of injured lymphatic ducts has recently been described in treating lymphatic leaks in the thorax and abdomen. 18
Lymphatic embolisation was indicated when chyle drainage exceeded 1000 ml in 24 h or >300 ml/24 h for over 1 week. The indication for non-conservative treatment varies widely among publications, ranging from chyle drainage of >250 ml/24 h to >1000 ml/24 h. 2,4,5,7 Despite the general agreement that significant loss of chyle over a sustained period results in nutritional and immunological consequences, surgical intervention—which represented non-conservative treatment for a long time—carries its own risk owing to its invasive nature. Considering the indication of non-conservative treatment is largely based on surgical literature, the invasiveness of surgery may be the reason behind the lack of consensus on the threshold for non-conservative management. There may be a need to revise indications for non-conservative management to suit less-invasive strategies such as lymphangiography and thoracic duct embolisation. In patients with lymphorrhea from surgical wounds, the amount of chyle leak could not be accurately measured. Patients with lymphorrhea were treated due to the requirement for frequent changes of dressing for more than 1 week. In the literature, the threshold for non-conservative management of chyle lymphorrhea is comparatively lower due to the risk of wound dehiscence and infection when the leak persists. 25,26 Lastly, the patient with enlarging lymphocele had undergone repeated needle aspiration due to recurrent expansion of the lesion. Daily output was unknown at the time of referral because a catheter had never been placed until the patient underwent lymphangiography. Ultrasound assessment of the lymphocele revealed a short, prominent fistulous tract between the lymphocele and terminal thoracic duct, just above the lymphovenous junction. Considering potential complications from sclerosants such as alcohol being introduced into the systemic circulation, sclerotherapy was not considered. Instead, the terminal thoracic duct was embolised via retrograde access to interrupt chyle flow into the lymphocele.
This study possesses limitations related to its retrospective nature. A common strategy for conservative management, percutaneous intervention, and follow-up was not developed for this study. Another major limitation is the small sample size which owes to the low incidence of chyle leak after neck surgery and the rarity of cases that require interventional or surgical treatment.
In conclusion, lymphatic embolisation seems effective and safe in managing chyle leaks after neck surgery. Lymphangiography allowed for the categorisation of chyle leaks according to their location. Post-embolisation patency of the thoracic duct may be preserved in chyle leaks that do not directly involve the thoracic duct.
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