334
CASE REPORT THOMPSON ET AL ROBOTIC LIGATION TO TREAT CHYLOTHORAX
FEATURE ARTICLES
mains to be clarified, but some explanations have been investigated. The long-term inflammation causes metaplasia in the bronchial and pleural epithelium and the epithelium of the skin margins of the tract. Subsequently, metaplastic epithelium extends into the chest cavity where cell proliferation and malignant degeneration occur [2]. The other explanation relies on the observation that patients in whom artificial therapeutic pneumothorax was used for tuberculosis presented with carcinoma many years after the treatment [3–5]. This suggests that exposure of the thoracic cavity to the environment causes degenerative changes resulting in malignancy [3, 4]. The reports indicate that longstanding chronic irritation may result in carcinoma in the range of 11 to 51 years [1– 6]. In the present case, the patient had undergone a left pneumonectomy 25 years ago, but no pleural disease was noted in her medical record and no complication occurred postoperatively. The patient underwent a routine follow-up for 8 years postoperatively and survived for 25 years without any complaints. Thus, in this patient, regarding the mechanisms for cancer development, exposure of the cavity seems to be a risk factor rather than a chronic irratation. It seems that onset of the symptoms was caused by the large mass. Initially the mass probably resulted in a microfistula causing an intrapleural infection and subsequently a large fistula developed. It has been reported that bronchopleural fistula occurs many years after pneumonectomy; however, 25 years remains an unusually long interval. Malignancy originating from a bronchial stump may well be a reason for bronchopleural fistula, which can be easily diagnosed with bronchoscopy. Emergency chest tube insertion is a basic approach for empyema and bronchopleural fistula to prevent contralateral aspiration; however, the exceptional history of this patient prompted us to obtain a computed tomographic scan prior to drainage. Further evaluation was performed in the operating room because simple drainage was not considered to be adequate. In conclusion, surgeons should note that malignant degenerations may develop in the thoracic cavity after
Ann Thorac Surg 2008;85:334 – 6
Fig 3. Macroscopic view of the tumor.
pneumonectomy in patients who have a long life expectancy. Postoperative chest x-ray films obtained many years after pneumonectomy must also be reviewed in this regard.
References 1. Cattaneo SM. Carcinoma of the chest wall complicating chronically draining empyema. Chest 1973;64:673– 6. 2. Deaton WR. Carcinoma arising in chronic empyema cavity: Case report with review of the literature. Chest 1962;42: 563– 6. 3. Rena O, Casadio C, Maggi G. Primitive squamous-cell carcinoma after extrapleural pneumothorax for active tuberculosis. Eur J Thorac Cardiovasc Surg 2001;19:92–5. 4. Gross P, Harley A. Asbestos-induced intrathoracic tissue reactions. Arch Pathol 1973;96:245–50. 5. Prabhakar G, Mitchell IM, Guha T, et al. Squamous cell carcinoma of the pleura following bronchopleural fistula. Thorax 1989;44:1053– 4. 6. Ruttner JR, Heinzl S. Squamous cell carcinoma of the pleura. Thorax 1977;32:497–500.
Treatment of Chylothorax by Robotic Thoracic Duct Ligation Kari J. Thompson, MD, Kemp H. Kernstine, MD, PhD, Frederic W. Grannis, Jr, MD, Pablo Mojica, MD, and Andres Falabella, MD City of Hope, Duarte, California
This is the first report describing the use of robotic technology for the treatment of chylothorax. We present Accepted for publication April 26, 2007.
Fig 2. Thorax computed tomographic scan reveals the tumor in the thoracic cavity. © 2008 by The Society of Thoracic Surgeons Published by Elsevier Inc
Address correspondence to Dr Kernstine, City of Hope, 1500 East Duarte Rd, Duarte, CA 91010; e-mail:
[email protected].
0003-4975/08/$34.00 doi:10.1016/j.athoracsur.2007.04.109
Ann Thorac Surg 2008;85:334 – 6
CASE REPORT THOMPSON ET AL ROBOTIC LIGATION TO TREAT CHYLOTHORAX
335
C
hylothorax secondary to thoracic duct trauma occurs in post-thoracotomy patients with a frequency of 0.25% to 0.5% [1]. Risk factors include extensive mediastinal dissection, mediastinal radiation therapy, or mediastinal neoplasia. There are 40% to 60% of patients who exhibit anatomical variability of the thoracic duct, making it challenging to avoid during surgical dissection. Potential methods to address iatrogenic chylothorax include observation, mechanical drainage, low fat diet with or without medium chain triglycerides supplementation, pleurodesis, oversewing of the thoracic duct injury, lymphatic ligation, and prophylactic thoracic duct ligation [2– 4]. Due to malnutrition secondary to protein and fatty acid loss, immunosuppression from lymphocytes loss, and respiratory compromise from atelectasis, treatment of chylothorax is indicated. Minimally invasive treatment of traumatic or iatrogenic chylothorax has been described [5–7]. Ligation of the thoracic duct at the entrance to the posterior mediastinum is highly successful; however, port placement and visibility in the inferior posterior sulcus can be cumbersome. Given our prior experience with robotic esophagectomy, we speculated that a robotic approach would decrease operative time and morbidity. Threedimensional visibility and numerous degrees of freedom allow precise ligation. We present a case of post-thoracotomy chylothorax refractory to conservative management. Robotic thoracic duct ligation was performed with immediate arrest of chylous drainage. This is the first report describing the use of robotic technology for treatment of chylothorax. The patient is a 22-year-old, nonsmoking, college student with a history of right testicular mixed embryonal cell and semi-nomatous germ cell tumor with metastatic disease to the retroperitoneum, liver, mediastinum, and both lungs. After right orchiectomy and 11 cycles of chemotherapeutic regimens consisting of platinum, vincristine, methotrexate, bleomycin, actinomycin, cyclophosphamide, and etoposide (POMB-ACE), a new lesion developed and the normalized markers (ie, beta HCG and alpha-fetoprotein) began to rise. The patient was then treated with gemcitabine, oxaliplatin, and Taxol (paclitaxel; Bristol-Myers Squibb, Princeton, NJ) (GOT) for eight cycles. After treatment, a single liver nodule persisted. It was resected by a laparotomy. Histologically the nodule was comprised of necrotic tumor. Lesions subsequently appeared in both lungs and markers rose again. He was then treated with vinblastine, ifosfamide,
Fig 1. After double lumen intubation, the patient is positioned on the left side, somewhat down and semiprone. The robotic chassis is brought over the patient’s back, left side. Four ports are placed: three for the robotic arms (circled) and one accessory 12-mm port (short line). The rightward robotic (8 mm) port is placed at the fifth intercostal space adjacent to the anterior aspect of the scapular tip, posterior to the posterior axillary line. Along the same linear plane, approximately 12 to 14 cm away, the 12-cm port is placed for the robotic videoscope and the third robotic port (8 mm) is placed 12 to 14 cm away along the same place as the first two ports. The final port is placed 14 cm anterior to the plane of the first three ports. The fan retractor and suture are passed through it.
and cisplatin (VEIP) for four cycles and markers normalized. Two months later, markers rose to alphafetoprotein of 25.1 nG/mL (nL ⱕ 10.8), lactate dehydrogenase 547 U/L (297–537 nL) and a beta human chorionic gonadotropin 129.7 (nL ⬍ 40). A single 2.5-cm left lower lobe pulmonary nodule was found on computed tomography. Although the prognosis was understood to be poor, surgical salvage was attempted. Under single lung ventilation, an exploratory thoracoscopy was performed. The pulmonary nodule was not visualized. A left-sided thoracotomy was performed. Enlarged, matted lymph nodes were resected along with the left lower lobe (R2 resection). Immediate pathologic examination of the mass demonstrated persistent embryonal cell cancer with extensive nodal involvement in nine of ten perihilar nodes and three of four aortopulmonary window nodes. Drains were placed and the chest was closed. On postoperative day 5, chest tube drainage doubled and became chylous with a triglyceride level of 170 mg/dL, total protein of 2.4 G/dL, and glucose of 66 mg/dL. The patient was placed on a low-fat diet, and chest tube suction was decreased to ⫺10 cm H2O. Chest tube drainage increased from 560 mL/day to 1100 mL per day. Pleural effusion was not seen on the follow-up chest x-ray film in spite of the reduced suction. A central venous line was placed, total parenteral nutrition was initiated, and oral feeding was discontinued. For the next 2 days, daily thoracic output increased to 1.6 L/day. After consideration of all options, on postoperative day 7, a right-sided robotic approach with single lung ventilation was performed. With the patient in a nearly prone and slightly reversed Trendelenburg position, four port sites were made (two for the robotic arms, one for the robotic videoscope, and one for a fan retractor and passage of suture) (Fig 1). With 15 mm Hg of CO2 insufflation there was good visualization of the most
FEATURE ARTICLES
a 22-year-old with mixed embryonal cell and seminoma germ cell cancer refractory to medical and surgical treatment. The patient had rising markers and a growing left lower lung lobe metastasis. After left lower lobectomy, left-sided chylothorax developed. Conservative management failed, and a robotic right-sided thoracic duct ligation was performed. Other treatment options are reviewed. (Ann Thorac Surg 2008;85:334 – 6) © 2008 by The Society of Thoracic Surgeons
336
CASE REPORT LIMMER ET AL PULMONARY LYMPHANGIOMA
inferior aspect of the posterior sulcus. Just cephalad to the diaphragm, the posterior mediastinal tissue between the descending aorta and azygous vein was triple ligated with No. 0 Ethibond (Ethicon Inc, Johnson and Johnson, Cincinnati, OH) in a figure eight fashion. Two No. 19 round Blake thoracostomy drains (Ethicon Inc) were placed. On postoperative day 1, output from the left chest tube decreased to 200 mL of clear fluid. Output from the right chest drains was minimal. Over the next 2 days the drains were removed and the patient was discharged home. Chest x-ray films 2 weeks later demonstrated no evidence of effusion.
Comment
FEATURE ARTICLES
Chylothorax represents a significant health risk increasing morbidity and mortality as well as healthcare costs. To guide treatment, algorithms encompassing medical and surgical treatment options have been established. In this immune-compromised patient, persistent chylothorax would have further lengthened hospitalization, increased the likelihood of postoperative complications (including wound infection, bronchial stump disruption, and pneumonia), and delayed additional chemotherapy. Ligation of the thoracic duct was considered preferable to pleurodesis, as the latter has inconsistent efficacy especially in patients with large volumes of effusion. Due to the degree of prior mediastinal dissection, there was concern that it would have been difficult to identify the lymphatic injury, potentially creating injury to adjacent, uninvolved structures. Right-sided thoracoscopy was also considered, but the small size of the inferior posterior sulcus, especially in the heavier patient, would have made accurate deep suture placement technically difficult. Our choice was to ligate the thoracic duct where it enters the mediastinum from the retroperitoneum, between the ascending aorta and the azygous vein [8]. We commonly perform this technique robotically during our robotic esophagectomy and resection of posterior mediastinal masses [9]. Docking times are typically approximately 10 minutes. In this case, total operative time was 40 minutes. Robotic surgery allowed enhanced visibility in a small space, facilitated suture placement, and minimized patient morbidity. Robotic thoracic duct ligation is an attractive treatment modality for treatment of persistent chylothorax.
References 1. Cerfolio RJ, Allen MS, Deschamps C, et al. Postoperative chylothorax. J Thorac Cardiovasc Surg 1996;112:1361– 6. 2. Cope C, Kaiser LR. Management of unremitting chylothorax by percutaneous embolization and blockage of retroperitoneal lymphatic vessels in 42 patients. J Vasc Interv Radiol 2002;13:1139 – 48. 3. Valentine VG, Faffin TA. The management of chylothorax. Chest 1992;102:586 –91. 4. Ferguson MK, Little AG, Skinner DB. Current concepts in the management of postoperative chylothorax. Ann Thorac Surg 1985;40:542–5.
Ann Thorac Surg 2008;85:336 –9
5. Buchan KG, Hosseinpour AR, Ritchie AJ. Thoracoscopic thoracic duct ligation for traumatic chylothorax. Ann Thorac Surg 2001;72:1366 –7. 6. Pimpec-Barthees FL, D’Attellis N, Dujon A, Legman P, Riquet M. Chylothorax complicating pulmonary resection. Ann Thorac Surg 2002;73:1714 –9. 7. Wuring PN, Hollaus PH, Ohtsuka T, Flege JB, Wolf RK. Thoracoscopic direct clipping of the thoracic duct for chylopericardium and chylothorax. Ann Thorac Surg 2000;70: 1662–5. 8. Kausel HW, Reeve TS, Stein AA, et al. Anatomic and pathologic studies of the thoracic duct. J Thorac Surg 1957;34:631. 9. Kernstine KH, DeArmond DT, Karimi M, et al. The robotic, 2-stage, 3-field esophagolymphadenectomy. J Thorac Cardiovasc Surg 2004;127:1847–9.
Pulmonary Lymphangioma Stefan Limmer, MD, Manuela Krokowski, MD, and Peter Kujath, MD Departments of Surgery and Pathology, University Hospital of Schleswig-Holstein, Lübeck, Germany
We report on the very rare case of a 49-year-old man with a large solitary pulmonary lymphangioma. Rapid growth of the tumor led to dyspnea and pain. A chest roentgenogram and computed tomography scan revealed a large 18 ⴛ 12-cm space-occupying cystic lesion in the posterior mediastinum. The tumor was resected by lateral thoracotomy. Histopathology revealed a pulmonary cystic lymphangioma. (Ann Thorac Surg 2008;85:336 –9) © 2008 by The Society of Thoracic Surgeons
L
ymphangioma is a congenital lymphatic malformation that develops from a defective bud formation [1]. A distinction is drawn between pulmonary lymphangioma (local defect), pulmonary lymphangiomatosis (multifocal defects), congenital lymphangiectasia (pathologically dilated lymphatic vessels in their anatomical position), mixed vascular and lymphatic angioma, and combined defects such as lymphangioleiomyomatosis. Histomorphologically, these lesions show multiple cystic cavities lined with endothelium. We report the case of a very rare large solitary cystic pulmonary lymphangioma. A 49-year-old man was admitted to the thoracic surgery unit of the Schleswig-Holstein University Clinic, Campus Lübeck, for further investigation of a space-occupying lesion in the posterior mediastinum. He had complained for 3 weeks of retrosternal pain, intermittent dyspnea, and right chest pain on deep inspiration. There was no dysphagia. The patient had a smoking history of 25 pack-years. The physical examination and laboratory variables showed physiologic values within normal reference ranges. His lung function was normal for age, with a forced expiratory volume in 1 second (FEV1) of 3.13l, and Accepted for publication July 10, 2007. Address correspondence to Dr Limmer, Department of Surgery, University Hospital of Schleswig-Holstein, Campus Lübeck, Ratzeburger Allee 160, Lübeck, 23538, Germany; e-mail:
[email protected].