BRIEF REPORT

Percutaneous Transcervical Thoracic Duct Embolization for Treatment of a Cervical Lymphocele Following Anterior Spinal Fusion: A Case Report Patrick S. Warren, MD, Mark J. Hogan, MD, and William E. Shiels, DO ABSTRACT Thoracic duct injury is an uncommon complication of neck dissection and cervical spinal surgery that is associated with significant morbidity. The authors describe an unusual case of thoracic duct injury during anterior spinal fusion resulting in a large prevertebral lymphocele presenting with dysphagia, respiratory distress, and chyloptysis. Surgical closure of the lymphocele was unsuccessful, and percutaneous drainage and sclerotherapy was performed. A large thoracic duct branch communicating with the lymphocele became evident during sclerotherapy, and embolization of the duct was performed via a percutaneous transcervical approach. Symptoms immediately resolved, and the patient remained asymptomatic at 6-month follow-up.

ABBREVIATION PTDE = percutaneous thoracic duct embolization

Chylous leaks are uncommon entities resulting from disruption of lymphatic channels with leakage of intestinal lymph (ie, chyle) outside of the lymphatic system. A chylous leak may occur anywhere along the course of the thoracic duct, cisterna chili, or intestinal lymphatic ducts. The etiology of lymphatic injury may be traumatic (eg, surgical) or nontraumatic (eg, idiopathic, malignant), and the presentation and clinical course are highly variable and relate to the anatomic location of the disruption (1). Clinical presentations include chylous ascites, chylothorax, chylopericardium, chyloptysis, chyluria, lymphocele formation, and chylous wound leaks (1). The present case report details the presentation and management of a postsurgical chylous leak related to cervical discectomy and anterior spinal fusion that was successfully treated via percutaneous embolization of a thoracic duct branch via a transcervical approach.

From the Department of Radiology, Nationwide Children’s Hospital, 700 Children’s Dr., Columbus, OH 43205. Received January 14, 2013; final revision received March 27, 2013; accepted April 2, 2013. Address correspondence to P.S.W.; E-mail: [email protected] None of the authors have identified a conflict of interest. & SIR, 2013 J Vasc Interv Radiol 2013; 24:1901–1905 http://dx.doi.org/10.1016/j.jvir.2013.04.009

CASE REPORT Institutional review board approval for case reports is not required by our institution. A 58-year-old woman with advanced cervical degenerative disease underwent cervical discectomies with anterior spinal fusion at the C3 through C7 vertebral levels performed through a right-sided approach. One week after surgery, chyloptysis, dysphagia, and respiratory distress developed secondary to the formation of a large prevertebral fluid collection first identified on a lateral neck radiograph (Fig 1). Magnetic resonance (MR) imaging of the neck confirmed the presence of a unilocular prevertebral fluid collection measuring 10 cm  7.5 cm  3 cm bordered laterally by the internal jugular veins and anteriorly by the trachea, esophagus, and thyroid gland (Fig 2). The imaging findings suggested a postsurgical lymphocele with presumed thoracic duct injury, and surgical exploration was performed 2 weeks after spinal fusion in an attempt to repair the leak. Surgical repair was unsuccessful, and the patient was referred to interventional radiology for percutaneous management. Symptoms included expectoration of 15 mL/h of chylous fluid, dysphagia, and dyspnea, and the patient exhibited significant respiratory distress exacerbated by neck extension and supine positioning. Esophagography showed marked prevertebral soft-tissue fullness adjacent to the spinal fusion hardware, with anterior displacement of the esophagus and trachea. Given her respiratory distress, urgent percutaneous aspiration of

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Figure 1. Lateral neck radiograph shows C3 through C7 cervical discectomies and anterior cervical fusion hardware. There is an associated large prevertebral soft-tissue mass (arrow) with anterior displacement of the trachea and esophagus.

Figure 3. Oblique spot fluoroscopic image obtained following iodinated contrast agent study of the cervical lymphocele through a 12-F drainage catheter demonstrates the large dimensions of the cavity.

Figure 2. Axial T2-weighted MR image of the neck shows a prevertebral fluid collection (asterisk) displacing the cervical trachea and esophagus anteriorly and to the left.

the collection was performed under ultrasound (US) guidance. Access was obtained from a right-sided approach anterior to the carotid sheath and posterior to the right lobe of the thyroid gland. A total of 65 mL of milky, opaque fluid was aspirated, with immediate relief of the patient’s respiratory distress. A 12-F pigtail drainage catheter (Boston Scientific, Natick,

Massachusetts) was placed and connected to bulb suction (Fig 3). A total of three catheter-based sclerotherapy treatments were performed at 2-week intervals. The sclerotherapy protocol consisted of transcatheter administration of 15 mL of 3% sodium tetradecyl sulfate (Bioniche, Lake Forest, Illinois) allowed to dwell for 2 minutes, followed by aspiration and subsequent administration of 10 mL of 98% dehydrated alcohol (Akorn, Lake Forest, Illinois), which was allowed to dwell for 20 minutes before aspiration. Each treatment resulted in progressive improvement of symptoms and decreased dimensions of the cavity on sonography, although the volume of chylous drainage remained unchanged at 75–100 mL/d and chyloptysis persisted. At the first treatment, an iodinated contrast agent study of the collection revealed a 3-mm-diameter lymphatic channel arising from the inferior aspect of the cavity. This channel traveled caudally along the anterior thoracic spine, appearing to communicate with a normal left thoracic duct (Fig 4). The lymphatic channel remained open despite sclerotherapy and was believed to be responsible for persistent chylous output and incomplete closure of the cavity. Percutaneous transcatheter embolization of this thoracic duct branch was planned via access

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Figure 4. (a) Anteroposterior and (b) oblique images obtained following continued contrast agent injection into the lymphocele show contrast agent opacification of a right lymphatic duct (black arrow) communicating with a plexus of thoracic duct channels (asterisk) located left of midline and a normal-appearing left thoracic duct (white arrow). The left thoracic duct appears to terminate at a normal left venolymphatic junction (white arrowhead, a).

through the lymphocele cavity with use of the existing drainage tract. The indwelling drainage catheter was exchanged over a guide wire for a 6-F hemostatic valve sheath. A 4-F C2 glide catheter (Terumo, Somerset, New Jersey) was used to cannulate the origin of the duct. A 1.7-F Echelon microcatheter (Covidien, Plymouth, Minnesota) was advanced centrally into the duct, and transcatheter embolization with n-butyl cyanoacrylate glue (Codman and Shurtleff, Raynham, Massachusetts) and Lipiodol (Guerbet, Roissy, France) combined in a 1:1 ratio was performed (Fig 5). Following embolization, a 12-F drainage catheter was replaced into the residual cavity. Immediately after the procedure, chylous drainage from the cavity ceased, and the drain was removed 48 hours later. Chyloptysis resolved immediately after embolization. At follow-up clinic visits at 1, 3, and 6 months, the patient continued to report absence of chyloptysis and no return of dysphagia or respiratory distress. US of the neck at each clinic encounter showed no evidence of residual or recurrent lymphocele.

DISCUSSION Surgical thoracic duct injuries, although uncommon, represent serious complications with considerable morbidity related to fluid accumulation/mass effect and loss of protein and electrolyte-rich chyle. If the volume of the leak exceeds

1 L/d, electrolyte disturbance will occur in 1–2 days and prolonged leaks will eventually result in immunodeficiency and malnutrition (2,3). Most chylous leaks are iatrogenic in nature, with the majority of cases resulting from abdominal or thoracic surgery (4). Thoracic duct injury during head and neck surgery is less frequent and generally seen following left neck dissection, with an estimated incidence of 3% (5). Treatment traditionally consists of conservative measures intended to decrease chyle production, including dietary modification (eg, nonfat diet, total parenteral nutrition) with or without pharmacologic therapy (eg, somatostatin or somatostatin analogues), or more invasive therapies such as surgical ligation of the thoracic duct or percutaneous thoracic duct embolization (PTDE) (2). Before the introduction of PTDE by Cope et al (7), surgical ligation of the duct was considered the first-line intervention, but morbidity and mortality rates of 38% and 2%–16%, respectively, and failure rates as high as 10% have led to the acceptance of PTDE as the new first-line intervention (7–10). Since the introduction of PTDE, there have been no reports of mortality associated with the procedure, and morbidity rates appear very low (2%) (11). In addition to reduced morbidity and mortality, PTDE also has the advantage of being minimally invasive and providing the ability to identify chyle leaks and variations in thoracic duct anatomy (12). The introduction of US-guided intranodal lymphangiography further reduced immediate complications associated with pedal

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Figure 5. Digitally subtracted image obtained during coaxial cannulation of the right lymphatic duct with a 4-F directional catheter and 1.7-F microcatheter. From this position, the duct was embolized with n-butyl cyanoacrylate glue (not shown).

lymphangiography as well as improved technical success rates and shorted procedural times as a result of decreased technical complexity (13). Delayed complications of thoracic duct embolization relate to the obstruction of flow of intestinal chyle, which can result in lower-extremity lymphedema, chylous ascites, and chronic diarrhea, and were reported in 14.3% of patients in a retrospective study of 49 patients by Laslett et al (14). There are multiple aspects of the case of thoracic duct injury and subsequent PTDE described here that are unusual and notable. Thoracic duct injury following anterior cervical spinal fusion via a right anterior approach is uncommon (6). There have been numerous reports documenting chyle leak following neck dissection, particularly left neck dissection, related to injury of the terminal arch of the thoracic duct as it approaches the venolymphatic junction, typically located at the confluence of the left subclavian and internal jugular veins (6,15). The configuration of the lymphatic system depicted in anatomic texts showing a single dominant thoracic duct draining the lower extremities, abdomen, chest, left upper extremity, and left neck occurs in only approximately 63% of patients (15). Much like the venous system, the lymphatic system develops embryologically from paired bilateral lymphatic trunks connected by numerous collateral anastomoses, most of

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which regress, leaving a single definitive thoracic duct. However, there is a high degree of variability in the configuration of the thoracic duct related to differing patterns of formation and regression of the embryonic lymphatic channels (15). Variations can be seen at the termination of the thoracic duct, its course, the number of ducts, and the location of its tributaries. The course of the thoracic duct can be predominantly left-sided, rightsided, or bilateral, in the case of a duplicated thoracic duct (1). Based on our imaging, it is unclear which particular variant was present in the patient described here, but we suspect bilateral thoracic ducts or bilateral outflow of a single thoracic duct. PTDE was not initially considered for this patient, as management first involved decompression of the lymphocele to alleviate respiratory compromise, with sclerotherapy then planned for more definitive treatment. It was our hope that sclerotherapy could obliterate the lymphocele and cease chylous output, and although partial success was achieved with decreased volume of the lymphocele, persistent high chylous drainage output necessitated further intervention. During the first sclerotherapy treatment, the communication with the thoracic duct was identified, and it was our hope that the cavity and duct could both be successfully sclerosed. It became apparent that the high output of the leak limited the effectiveness of transcatheter sclerotherapy, and embolization of this duct was considered. The residual lymphocele was capacious enough to accommodate a 4-F directional catheter, which allowed manipulation of a microcatheter into the injured lymphatic channel, through which selective embolization of the duct could be safely performed. This approach proved successful and eliminated the need to perform pedal or intranodal lymphangiography to cannulate the cisterna chyli for conventional PTDE as originally described by Cope et al (7). In conclusion, cervical thoracic duct injuries are rare, and successful identification and closure of the injured duct may be challenging. The case described here provided the opportunity to embolize the injured duct through a cervical lymphocele, minimizing the risk to the patient from more invasive approaches. Although few cases of thoracic duct injury are likely to be amenable to this approach, the treatment algorithm for chyle leaks into a confined space such as this could include percutaneous access with contrast agent interrogation of the space in an attempt to visualize and embolize the injured duct.

REFERENCES 1. Chen E, Itkin M. Thoracic duct embolization for chylous leaks. Semin Intervent Radiol 2011; 28:63–74. 2. Brennan PA, Blythe JN, Herd MK, Habib A, Anand R. The contemporary management of chyle leak following cervical thoracic duct damage. Br J Oral Maxillofac Surg 2012; 50:197–201.

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3. Marts B, Naunheim K, Fiore A, Pennington D. Conservative versus surgical management of chylothorax. Am J Surg 1992; 164:532–534. 4. Doerr CH, Allen MS, Nicholas FC III, Ryu JH. Etiology of chylothorax in 203 patients. Mayo Clin Proc 2005; 80:867–870. 5. de Gier HH, Balm AJ, Bruning PF, Gregor RT, Hilgers FJ. Systematic approach to the treatment of chylous leakage after neck dissection. Head Neck 1996; 18:347–357. 6. Hart AK, Grienwald JH, Shaffrey CI, Postma GN. Thoracic duct injury during anterior spinal discectomy: a rare complication. J Neurosurg 1998; 88:151–154. 7. Cope C, Salem R, Kaiser LR. Management of chylothorax by percutaneous catheterization and embolization of the thoracic duct: prospective trial. J Vasc Interv Radiol 1999; 10:1248–1254. 8. Merigliano S, Molena D, Ruol A, et al. Chylothorax complicating esophagectomy for cancer: a plea for early thoracic duct ligation. J Thorac Cardiovasc Surg 2000; 119:453–457.

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9. Cerfolio R, Allen M, Deschamps C, Trastek V, Pairolero P. Postoperative chylothorax. J Thorac Cardiovasc Surg 1996; 112:1361–1365. 10. Sieczka E, Harvey J. Early thoracic duct ligation for postoperative chylothorax. J Surg Oncol 1996; 61:56–60. 11. Marcon F, Irani K, Aquino T, Saunders J, Gouge T, Melis M. Percutaneous treatment of thoracic duct injuries. Surg Endosc 2011; 25:2844–2848. 12. Itkin M, Chen E. Thoracic duct embolization. Semin Intervent Radiol 2011; 29:261–266. 13. Nadolski G, Itkin M. Feasibility of ultrasound-guided intranodal lymphangiogram for thoracic duct embolization. J Vasc Interv Radiol 2012; 23: 613–616. 14. Laslett D, Trerotola SO, Itkin M. Delayed complications following technically successful thoracic duct embolization. J Vasc Interv Radiol 2012; 23:76–79. 15. Hematti H, Mehran R. Anatomy of the thoracic duct. Thorac Surg Clin 2011; 21:229–238.

Percutaneous transcervical thoracic duct embolization for treatment of a cervical lymphocele following anterior spinal fusion: a case report.

Thoracic duct injury is an uncommon complication of neck dissection and cervical spinal surgery that is associated with significant morbidity. The aut...
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