Bilateral chylothorax following left supraclavicular lymph node dissection for breast cancer: one case report and literature review

Abstract

Chylothorax is a rare complication of neck dissection, and bilateral chylothorax is even rarer. However, both are potentially serious and sometimes life-threatening, especially those that are associated with left neck dissection for head and neck neoplasms. We report one case of bilateral chylothorax following left supraclavicular dissection for breast cancer. This case was treated successfully with a new conservative management approach.

Isolated regional nodal recurrences after curative treatment of breast cancer are rare, especially in supraclavicular nodes[1]^,[2]. Clinical outcomes are similar for patients with ipsilateral supraclavicular lymph node metastases at first presentation and for patients with recurrent ipsilateral supraclavicular lymph node metastases[3]^,[4]. Per the last accepted staging system for breast cancer[5], ipsilateral supraclavicular lymph node metastases is classified as regional disease (stage IIIC); but the survival rate was lower in patients with ipsilateral supraclavicular lymph node metastases than in patients with lower axillary or subclavian nodal involvement[6]. Nevertheless, patients with ipsilateral supraclavicular lymph node metastases who were treated with surgery or radiotherapy and achieved good neck control were reported to achieve better survival than those for whom surgical treatment or irradiation did not result in good local control[6]^–[8]. Thus, neck dissection is sometimes necessary for breast cancer patients with ipsilateral supraclavicular lymph node metastases.

Chylous leakage is a common complication following neck dissection. However, postoperative chylothorax is uncommon, and bilateral chylothorax is even rarer. This condition may cause severe respiratory, metabolic, and immunologic dysfunctions and can even be fatal. Currently, there are no reported cases of bilateral chylothorax following neck dissection for breast cancer in the literature. Herein, we report a case of bilateral chylothorax following left supraclavicular dissection for breast cancer, which was treated successfully using a novel conservative management strategy.

Case Report

A 43-year-old woman presented with a 3-year history of a left-sided palpable breast mass. Physical examination revealed a 15 cm × 15 cm lump, fixed, invading the skin, accompanied with left supraclavicular lymphadenectasis. Results of chest X-ray, ventral ultrasonography, radioisotope bony scanning, and nuclear magnetic resonance imaging (MRI) of the head did not show distant metastasis. The pathologic diagnosis via core biopsy was adenocarcinoma, and clinical stage was IIIC (T4N3M0). The patient underwent 4 cycles of neoadjuvant chemotherapy, and therefore the mass was reduced to 25% of its original size (partial response). Radical mastectomy and left supraclavicular lymph node dissection were then performed. Chylous fluid output in the neck drainage tube was approximately 500 mL on the first day postoperation, but increased to 1,600 mL on the second day. An emergency operation was performed to ligate the fistula, and the leakage was ceased. Nevertheless, on the fourth day postoperation, the patient experienced dyspnea and chest discomfort. Physical examination revealed no neck edema or swelling, but bilateral breath sounds were diminished on auscultation. Chest X-ray, which did not show obvious abnormalities preoperatively (Figure 1A), revealed medium, bilateral pleural effusions (Figure 1B). Diagnostic thoracentesis was performed and milky fluid was aspirated from both sides. Biochemical analysis confirmed bilateral chylothorax.

Figure 1. Bilateral chylothorax occured after left supraclavicular dissection.

A, no obvious abnormalities are present on the preoperative chest X-ray. B, on the fourth day postoperation, chest X-ray shows medium, bilateral pleural effusions, accompanied with dyspnea and chest discomfort. Bilateral breath sounds were diminished upon auscultation. C, after conservative management, a repeated chest X-ray shows no evidence of residual effusions. These results suggest that conservative treatment of chylous leakage is effective.

We implemented a conservative management approach, which involved insertion of bilateral pigtail catheters into the thoracic cavity, total parenteral nutrition with total enteric rest, and administration of somatostatin (250 µg/h) by continuous intravenous drip on the basis of the review of literature. On the 12th day postoperation, the patient achieved complete remission of dyspnea and chest discomfort; 970 mL of chyle-like liquid was aspirated intermittently from bilateral pleural spaces. A repeated chest X-ray showed no evidence of residual effusions (Figure 1C). Thus, the drainage tubes were removed, and the patient was started on a low fat and protein diet for 3 days before resuming a regular diet. On the 15th day post-operation, the patient had recovered completely.

Discussion

Bilateral chylothorax is a rare complication that results from injury to the thoracic duct or one of its branches following neck dissection. We searched the English-language literature through PubMed and found 26 cases of bilateral chylothorax reported between 1951 and 2011[9]^–[29]. We analyzed the primary diseases, treatment, and prognosis of bilateral chylothorax. The results showed that the majority of bilateral chylothorax cases followed neck dissection associated with head and neck cancers, such as thyroid cancer, laryngocarcinoma, and tongue cancer (Table 1). However, none of the reported cases described breast cancer as a cause for bilateral chylothorax.

Table 1. Cases of bilateral chylothorax following neck dissection reported in the literature.

Year	Authors (reference)	Disease	Neck dissection	Treatment	Prognosis
1951	Frazell et al.[9]	Carcinomacutaneumfaciale	Left	Conservative	Recovery
1976	Coates et al.[10]	Laryngocarcinoma	Left	Conservative	Recovery
1981	Saraceno et al.[11]	Carcinoma of the floor of the mouth	Left	Conservative	Recovery
1985	Har-EI et al.[12]	Thyroid cancer	Left	Conservative	Recovery
1985	Ng et al.[13]	Carcinoma of the floor of the mouth	Left	Conservative	Recovery
1988	Pace-Balzet et al.[14]	Adenolymphoma	Left	Conservative	Recovery
1989	Oi et al.[15]	Maxillary sinus carcinoma	Left	Conservative	Recovery
1992	Biurrun et al.[16]	Unclear	Unclear	Conservative	Recovery
1995	Jabbar et al.[17]	Thyroid cancer	Bilateral	Conservative	Recovery
2000	Gregor et al.[18]	Nasopharyngeal carcinoma	Right	Conservative	Recovery
2001	Al-Sebeih et al.[19]	Laryngocarcinoma	Bilateral	Conservative	Recovery
2001	Jortay et al.[20]	Unclear	Left	Conservative	Recovery
2003	Kamasaki et al.[21]	Tongue cancer	Bilateral	Conservative	Recovery
2004	Busquets et al.[22]	Adenolymphoma	Left	Surgery + conservative	Recovery
2006	Srikumar et al.[23]	Nasopharyngeal carcinoma	Left	Surgery + conservative	Recovery
2007	Tsukahara et al.[24]	Thyroid cancer	Left	Conservative	Recovery
		Tongue cancer	Left	Conservative	Recovery
		Laryngocarcinoma	Bilateral	Conservative	Recovery
2007	Bae et al.[25]	Thyroid cancer	Left	Conservative	Recovery
		Thyroid cancer	Left	Conservative	Recovery
2009	Han et al.[26]	Thyroid cancer	Left	Conservative	Recovery
2010	AL-Khudaris et al.[27]	Laryngocarcinoma	Bilateral	Conservative	Recovery
2011	Rodier et al.[28]	Thyroid cancer	Left	Conservative	Recovery
2011	Tian W et al.[29]	Thyroid cancer	Bilateral	Conservative	Recovery
		Thyroid cancer	Bilateral	Conservative	Recovery

The majority of injuries to the thoracic duct cause merely chylous leakage; chylothorax, especially bilateral, is rare. Milky fluid suggests a chylous effusion, but the absence of a milky appearance does not preclude chylothorax, especially if the patient is fasting or on a low-fat diet. Biochemical analysis of the fluid should be the initial diagnostic test, with a triglyceride concentration greater than 1,100 mg/L confirming the diagnosis[30]. When the triglyceride level is between 550 and 1,100 mg/L, a lipoprotein analysis is indicated to detect chylomicrons, whereas a triglyceride level less than 500 mg/L has no more than a 5% chance of being chylous[30]. Clinically, low-output chylothorax (<500 mL/day) could be treated using a conservative approach, such as drainage and a low-fat diet. High-output chylothorax (>500 mL/day) could cause a fluid and electrolyte imbalance and a loss of nutrients, and it can even be fatal. Thus, high-output chylothorax requires surgical intervention[31]. However, from the literature we reviewed, only 2 cases were treated with surgery[22]^,[23], whereas the remaining 24 cases were treated conservatively and recovered at length.

In this case, we performed an emergency operation to ligate the fistula on the second day postoperation, when the chylous leakage reached 1,600 mL in volume. Based on our clinical experience, postoperative adhesions develop by 3 days postoperation, making surgical intervention for chylous leakage a challenge. Although the surgery ceased the neck leakage, bilateral chylothorax was confirmed 4 days after operation. To treat this condition, we prefer to follow a conservative management plan rather than perform additional surgeries, unless the conservative approach is insufficient. Here, we chose nonsurgical treatments for the patient, including drainage, total parenteral nutrition with total enteric rest, continuous intravenous infusion of somatostatin (250 µg/h), and intermittent thoracocentesis for aspirating effusion, and the patient was cured.

Our report also supports that the use of somatostatin may be a new conservative therapeutic approach. The exact mechanism of action of this neurohormonal and panacrine agent remains unknown. It may reduce gastrointestinal chyle production by decreasing splanchnic blood flow and decreasing gastric, biliary, pancreatic, and intestinal secretions[28].

Taken together, our results and the results described in the previous literature suggest that conservative management is an effective treatment for bilateral chylothorax following neck dissection. Our report shows that the use of somatostatin may be a new conservative therapeutic approach.