Chylous fistula of the neck, which is related to the occurrence of chylothorax6), was reported in 2% of cases following neck dissection14), and chylothorax with chylous fistula of the neck is reported to be 0.2%6). Moreover, only 9 cases of chylothorax without chylous fistula were reported in the literature7–13), associated with head and neck cancer. The primary tumor included 4 cases of thyroid cancer, 2 cases of tongue cancer, 2 cases of laryngeal cancer, 1 case of lower gingival cancer, and 1 case of oral malignant melanoma (Table 1). Furthermore, 2 theories exist regarding the pathogenesis of chylothorax. First, chyle in the neck descends to the mediastinum through the intra-muscular space by compression of the neck. Then, chyle infiltrates to the lateral wall of the pleura, resulting in the accumulation chyle in the thoracic cavity15). Another theory is that ligation of the thoracic duct leads to elevation of its internal pressure causing rupture of the thoracic duct wall4). The normal range of internal pressure for the thoracic duct is reported as 10 to 25 mmHg; however, the internal pressure can rise to 50 mmHg due to duct closure16). Bilaterally affected chylothorax is related to the anatomical pathway of the thoracic duct. The thoracic duct originates at the cisterna chyli in front of L1 and L2 and ascends along with abdominal aorta into the thoracic cavity. At the level of T4, and T5, it turns left and ascends along with esophagus to finally join the left vein angle17). Although lymphangiography or lymph scintigraphy was not performed to avoid complications in this case, the point of thoracic duct rupture was estimated approximately at the level of T4 and T5, or the upper stream where chyle can spread to the bilatelal thoracic cavity.
The nature and color of the pleural fluid is essential for the diagnosis of chylothorax. A milky liquid is a typical finding in chylothorax; however, lymph fluid becomes transparent after fasting. Therefore, biochemical study to the pleural fluid is necessary. Over 110 mg/dL of triglyceride was the diagnostic finding for the chyle18), and if the triglyceride concentration was between 50 to 110 mg, the presence of cyromicron in the pleural fluid would be proof that it is chyle19). In the present case, the patient’s complaint of respiratory distress while in a supine position prompted us to suspect chylothorax. Moreover, the triglyceride concentration in pleural fluid was 705 mg/dL with intraoperative thoracic duct ligation; therefore, we easily arrived at the diagnosis of chylothorax.
Sella et al. reported that over 1500 mL of chyle, unchanged condition after concervative treatment for 14 days, and deterioration of nutrition by chyle exudation were the criteria for surgical intervention20). On the other hand, Robinson et al.21) suggested 500 to 1000 mL of chyle per day for 2 to 3 weeks for adults required surgical treatment. Although standardized criteria do not exist, these conditions support our selection of surgical intervention. Moreover, the American College of Radiology recommends lymphangiography and thoracic duct embolization as an effective and minimally invasive method for the treatment of chylothorax22).
Chylothorax is associated with increased mortality due to significant loss of essential proteins, immunoglobulins, fat, vitamins, electrolytes, and water23). Bolger et a al.24) reported that 50% of patients died if their treatment period was over 35 days. Also, the most frequent direct cause of death for chylothorax is infection25).
In this case, conservative treatment of low-fat diet improved the patient’s condition after 16 days of thoracentesis. Early diagnosis with adequate treatment is critically important for chylothorax following neck dissection.