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Head and neck

CASE REPORT

Direct puncture sclerotherapy of a thoracic duct cyst presenting as an enlarging left supraclavicular mass Yasha Kadkhodayan,1 Motoyo Yano,2 DeWitte T CrossIII2 1

Department of Interventional Neuroradiology, Consulting Radiologists Ltd, Abbott Northwestern Hospital, Minneapolis, Minnesota, USA 2 Mallinckrodt Institute of Radiology, Washington University School of Medicine, St. Louis, Missouri, USA Correspondence to Dr Yasha Kadkhodayan, yasha. [email protected] Republished with permissions from BMJ Case Reports Published 30 October 2013; doi:10.1136/bcr-2013-010844

ABSTRACT A 58-year-old woman presented with a palpable mass at the base of the left neck which she had first noticed 12 years previously, shortly after abdominal surgery. The mass had progressively enlarged, resulting in dysphagia, dyspnea and occasional pain. Imaging showed a septated but otherwise simple cystic mass extending into the mediastinum and containing lymphocytic fluid on aspiration. A diagnosis of the rare entity of a thoracic duct cyst with supraclavicular extension was made. The patient opted for percutaneous sclerotherapy of the lesion, which was performed using glacial acetic acid. This resulted in complete resolution of the mass with one treatment. After exclusion of other causes of cystic left supraclavicular masses including cystic neoplasms and pseudoaneurysms of the carotid or subclavian arteries, direct puncture sclerotherapy can be safe and effective.

BACKGROUND Thoracic duct cysts with supraclavicular extension are a rare entity with only several case reports in the literature, mostly describing diagnosis and surgical treatment.1 Sclerotherapy of similar lesions has only been described in a handful of cases using povidone iodine,2 OK-4323 4 and ethanol.5

CASE PRESENTATION

To cite: Kadkhodayan Y, Yano M, Cross DWT. J NeuroIntervent Surg Published Online First: [please include Day Month Year] doi:10.1136/ neurintsurg-2013-010844. rep

We present the case of a 58-year-old AfricanAmerican woman who first noted a mass at the base of the left neck 12 years previously, shortly after abdominal surgery. The mass had progressively enlarged, resulting in dysphagia to solids, dyspnea in the supine position and occasional pain and tenderness. There was no history of fever, bleeding, skin alteration over the mass or change in voice. Imaging indicated that the mass was entirely cystic and extended into the mediastinum. The lesion was aspirated twice; both times fluid reaccumulated in a relatively short time frame. As a separate problem, she had pulsatile tinnitus and decreased hearing on the right. Imaging revealed what was most likely a right glomus jugulare neoplasm. Her past medical history was significant only for the previous abdominal surgery; unfortunately, no record of exactly what was done was available. She also had a diagnosis of schizophrenia but had otherwise been in good health. She did not take any medications but smoked a pack of cigarettes daily. Heart disease and lung cancer ran in her family. On physical examination there was a fairly firm but compressible and non-tender mass of the left supraclavicular neck. There was no pulsation or

Kadkhodayan Y, et al. J NeuroIntervent Surg 2013;00:1–3. doi:10.1136/neurintsurg-2013-010844.rep

thrill associated with the mass and the skin overlying the mass was normal.

INVESTIGATIONS The lesion had been aspirated twice and cytology was reported as benign the first time, but with some atypical lymphatic cells on the second occasion (figure 1). The triglyceride level was 11 mg/ dL, lower than that of plasma. A recent CT showed a large cystic fluid attenuation mass extending from the left supraclavicular neck, beginning just below the left carotid bifurcation, displacing the left common carotid artery and internal jugular vein anteriorly, displacing the left subclavian artery posteriorly and displacing the trachea, esophagus and thyroid gland to the right. The mass extended parallel to the trachea and abutted the thoracic spine into the mediastinum where it extended below the carina to lie just anteromedial to the descending aorta and then disappeared just above the diaphragm. The supraclavicular portion on the left measured about 4 cm in diameter, bounded by a septum medially that separated it from another cystic compartment which measured about 4.5 cm in diameter at the sternoclavicular level. It became smaller inferiorly toward the carina, and was then separated by other septations from a cystic component extending along the aorta. There was no evident enhancement of the mass internally or at its periphery, and no hemorrhage or debris was seen within it.

DIFFERENTIAL DIAGNOSIS ▸ ▸ ▸ ▸

Lymphatic malformation Supraclavicular extension of a thoracic duct cyst Cystic lymphadenopathy or nerve sheath tumor Carotid or subclavian artery pseudoaneurysm

Figure 1

Sonographically guided needle aspiration. 1

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Head and neck TREATMENT The patient was referred by her otolaryngologist for percutaneous sclerotherapy in an attempt to reduce the size of the lesion, given her symptomatology and its recurrence following prior simple aspirations. After reviewing possible risks of the procedure including pain, bleeding, infection, damage to adjacent structures including nerve injury resulting in speech and swallowing difficulty, informed consent was obtained for the procedure, which was performed under general endotracheal anesthesia. With the patient in the supine position and the head turned to the right, the left neck was sterilely prepped and draped. The lesion was accessed percutaneously using an arterial micropuncture set and straw-colored fluid returned. Ultimately, a 4 Fr pigtail catheter was placed over a 0.035 inch J-wire and a total of 90 mL of fluid was aspirated. We then injected 50 mL of Optiray-320 followed by 20 mL of saline into the cyst under fluoroscopy. The resulting cystogram showed a large left lower neck cyst connected to the presumed dilated thoracic duct to the T7 level (figure 2). The injected contrast-saline mixture was aspirated. At this point we injected a total of 45 mL (half the volume of the originally aspirated fluid) of 50% glacial acetic acid, 25% contrast and 25% saline. This sclerosant mixture was allowed to indwell for 5 min and was then aspirated (figure 3). The pigtail catheter was removed, the left neck was cleansed with saline and a sterile bandage was applied. The patient was awakened from anesthesia in a stable condition. There were no complications.

OUTCOME AND FOLLOW-UP The patient reported complete resolution of the mass with no residual dysphagia or dyspnea on telephone follow-up at 6 weeks.

Figure 3

Aspiration of sclerosant.

DISCUSSION

Figure 2 Cystogram via 4 Fr pigtail catheter. 2

The thoracic duct is the largest lymphatic vessel in the body, originating from the cisterna chyli, which collects lymph from the intestinal and lumbar lymphatic trunks. The thoracic duct extends superiorly in the posterior mediastinum, collects thoracic lymph via posterior intercostal channels and empties into the junction of the left subclavian and jugular veins.6 There is considerable variability in lymphatic anatomy, particularly of the thoracic duct. Rarely, lymphoceles or cysts of the terminal part of the duct can develop and present as supraclavicular masses, of which there are a few case reports in the literature.7 Proposed theories as to the etiology include congenital weakness in the duct wall, an acquired inflammatory process, as well as postoperative or traumatic disruption of lymphatic drainage.8 The fluid within a lymphocele may be milky or straw-colored, as in this case. In a series of percutaneously treated abdominal and pelvic lymphoceles, 9 of 11 had clear straw-colored fluid rather than milky fluid and the location of the lymphocele seemed to matter.9 Lymph drained from the upper abdomen (ie, the gut) was of the milky variety, while that of the retroperitoneum and pelvis was clear or straw-colored. It may be that, as a result of our patient’s prior abdominal surgery after which she noted the mass, the milky lymph from the gut was disconnected from the thoracic duct collecting system. This also explains the low triglyceride level. While most reports say that triglycerides should be elevated in lymphatic fluid, lymphatic fluid from the lower body (excluding the gut) is an ultrafiltrate of plasma and may have lower triglyceride values.9 Kadkhodayan Y, et al. J NeuroIntervent Surg 2013;00:1–3. doi:10.1136/neurintsurg-2013-010844.rep

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Head and neck Traditional treatment of thoracic duct cysts has included observation or surgical resection in symptomatic cases. Surgery requires removal of the cyst and ligation of all lymphatics connected to the cyst.10 Depending on the size and location, there may be cosmetic implications with surgery or specific complications including chylothorax. There have been a few case reports of sclerotherapy for similar lesions using povidone-iodine,2 OK-4323 4 and ethanol5 which have all been used successfully. Successful treatment of lymphangiomas with acetic acid has also been reported. In a Korean series of 12 patients with lymphangiomas in various locations treated with acetic acid, complete resolution was achieved in eight patients and a reduction of >50% was achieved in three.11 Two had undergone failed sclerotherapy with a different agent. There were no serious complications. Acetic acid has also been used successfully in an abdominal lymphangioma that had failed multiple trials of ethanol.12 We were fortunate to replicate these results with acetic acid in a patient in whom follow-up in person would be difficult. Prior to sclerotherapy it is important to exclude other causes of cystic lesions in this location, such as cystic lymphadenopathy related to squamous cell carcinoma, papillary thyroid cancer, nerve sheath tumors of the carotid sheath or brachial plexus, lymphoma and metastatic disease from the chest, abdomen or pelvis. Carotid or subclavian pseudoaneuryms should also be excluded prior to instrumentation. If the diagnosis can be made with imaging and needle aspiration, percutaneous sclerotherapy of the rare supraclavicular thoracic duct cyst can be safe and effective.

Contributors YK and DTC performed the sclerotherapy. MY performed aspiration prior to sclerotherapy. YK drafted the manuscript. MY and DTC edited the manuscript for important intellectual content. YK is the guarantor of this report. Competing interests None. Patient consent Obtained. Provenance and peer review Not commissioned; externally peer reviewed.

REFERENCES 1

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Key messages

Lecanu JB, Gallas D, Biacabe B, et al. Lymphocele of the thoracic duct presenting as a left supraclavicular mass: a case report and review of the literature. Auris Nasus Larynx 2001;2013:275–7. Seelig MH, Klingler PJ, Oldenburg WA. Treatment of a postoperative cervical chylous lymphocele by percutaneous sclerosing with povidone-iodine. J Vasc Surg 1998;2013:1148–51. Miwa T, Tatsutomi S, Tsukatani T, et al. OK-432 therapy for a cervical thoracic duct cyst. Otolaryngol Head Neck Surg 2007;2013:852–3. Preuss SF, Wittekindt C, Beutner D, et al. Successful OK-432 therapy for a supraclavicular lymphocele: report of a case. Eur Arch Otorhinolaryngol 2006;2013:692–4. Dool JJ, de Bree R, van den Berg R, et al. Thoracic duct cyst: sclerotherapy as alternative for surgical treatment. Head Neck 2007;2013:292–5. Riquet M, Mordant P, Pricopi C, et al. [Anatomy, micro-anatomy and physiology of the lymphatics of the lungs and chest wall]. Rev Pneumol Clin 2013;2013:102–10. Mattila PS, Tarkkanen J, Mattila S. Thoracic duct cyst: a case report and review of 29 cases. Ann Otol Rhinol Laryngol 1999;2013:505–8. Offiah CE, Twigg S. Lymphocoele of the thoracic duct: a cause of left supraclavicular fossa. Br J Radiol 2011;2013:e27–30. White M, Mueller PR, Ferrucci JT Jr, et al. Percutaneous drainage of postoperative abdominal and pelvic lymphoceles. AJR Am J Roentgenol 1985;2013:1065–9. Gottwald F, Iro H, Finke C, et al. Thoracic duct cysts: a rare differential diagnosis. Otolaryngol Head Neck Surg 2005;2013:330–3. Won JH, Kim BM, Kim C-H, et al. Percutaneous sclerotherapy of lymphangiomas with acetic acid. J Vasc Interv Radiol J 2004;2013:595–600. Park SW, Cha IH, Kim KA, et al. Percutaneous sclerotherapy using acetic acid after failure of alcohol ablation in an intra-abdominal lymphangioma. Cardiovasc Intervent Radiol 2004;2013:285–7.

▸ Thoracic duct cyst is a rare but important entity in the differential diagnosis of a left supraclavicular mass. ▸ Percutaneous sclerotherapy of supraclavicular thoracic duct cysts can be safe and effective. ▸ It is important to exclude other causes of cystic lesions in this location prior to treatment.

Kadkhodayan Y, et al. J NeuroIntervent Surg 2013;00:1–3. doi:10.1136/neurintsurg-2013-010844.rep

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Direct puncture sclerotherapy of a thoracic duct cyst presenting as an enlarging left supraclavicular mass Yasha Kadkhodayan, Motoyo Yano and DeWitte T Cross III J NeuroIntervent Surg published online November 4, 2013

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Direct puncture sclerotherapy of a thoracic duct cyst presenting as an enlarging left supraclavicular mass.

A 58-year-old woman presented with a palpable mass at the base of the left neck which she had first noticed 12 years previously, shortly after abdomin...
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