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Ann Thorac Surg 1999;68:1053-1057
© 1999 The Society of Thoracic Surgeons
a Department of Surgery, St. Louis University Health Sciences Center, St. Louis, Missouri USA
b Departments of Surgery and Pediatrics, University of Virginia Health System, Charlottesville, Virginia, USA
Address reprint requests to Dr Rodgers, Department of Surgery, University of Virginia Health System, PO Box 10005, Charlottesville, VA 22906
e-mail: bmr{at}virginia.edu
Presented at the Forty-fifth Annual Meeting of the Southern Thoracic Surgical Association, Orlando, FL, Nov 12–14, 1998.
Abstract
Background. Traditional therapy for refractory chylothorax in the pediatric population has included pleurodesis and thoracic duct ligation. These procedures are associated with high morbidity and questionable success rates.
Methods. We retrospectively reviewed our experience with 15 patients who underwent treatment for chylous effusions using pleuroperitoneal shunts with exteriorized pump chambers. Mean patient age at time of shunt placement was 2.1 (0.1 to 11.5) years and the most common indication (7 of 15) was refractory chylothorax following surgical correction of congenital heart disease. Mean chylothorax duration before shunt placement was 76 (5 to 810) days and shunts were in place for an average of 104 (12 to 365) days. A total of 19 chylous effusions (pleural or pericardial) were treated with shunts.
Results. Nine of 11 right-sided chylothoraces, 5 of 6 left-sided chylothoraces, and 2 of 2 chylopericardia resolved with shunt therapy (84% total). Pleuroperitoneal shunting failed to clear the effusion in 3 children. There were six episodes of shunt malfunction that were repaired and two episodes of infection. Inguinal or umbilical hernia developed in 4 patients.
Conclusions. Externalized pleuroperitoneal shunting is a safe, effective, and minimally invasive treatment for children with refractory chylous effusions.
Persistent chylothorax in the pediatric patient, regardless of its etiology, has always presented a difficult clinical challenge. Prolonged severe chylothorax has been shown to result in compromise of the immune system, nutritional depletion, metabolic instability, electrolyte imbalance, impaired cardiac and respiratory functions, and death [1–4]. The morbidity and mortality resulting from chylothorax is a result of local, metabolic, and immunologic effects. Locally, chyle leakage into the pleural cavity causes compression of the ipsilateral lung. If the accumulation is sufficiently severe, mediastinal shift can occur, compromising the contralateral lung and impairing cardiac function. The metabolic and immunologic effects of persistent chylothorax are caused by the loss of chyle that is comprised mostly of T lymphocytes, chylomicrons from long-chain fatty acids, and an electrolytic composition similar to plasma.
Upon recognition of chylothorax, conservative therapy is indicated, consisting of restriction of oral fat intake to medium-chain triglycerides or use of total parenteral nutrition with medium-chain triglyceride supplementation. Drainage is accomplished by tube thoracostomy or serial thoracentesis. Conservative therapy often fails and is no longer indicated if the patient’s condition worsens or fails to improve after 2 weeks [2, 5–7].
Traditional therapy for chylothorax refractory to conservative management has included chemical or talc pleurodesis, pleurectomy, and thoracic duct ligation. Pleurodesis is a painful procedure and the long-term results are unreliable. Ligation of the thoracic duct using either video-assisted thoracoscopy (VATS) or a thoracotomy may fail as a result of anatomic variations of the thoracic duct or failure to identify the sites of chylous leakage [2, 8].
In 1982, Weese and Schouten [9] reported the successful use of pleuroperitoneal shunts in adults to palliate respiratory symptoms secondary to malignant pleural effusions. In 1983, Azizkhan and colleagues [10] described the treatment of refractory chylothorax with a pleuroperitoneal shunt in neonates.
The original version of the shunt was completely subcutaneous with a 1.5-mL pump chamber implanted along the costal margin. However, experience demonstrated that the small volume of the pump chamber necessitated frequent pumping, and that many children became irritable due to pain during chamber compression, discouraging use of this version of the shunt. The current version of the shunt (Denver Biomaterials, Inc, Golden, CO) consists of a flexible 2.5-mL pump chamber containing two one-way valves with 15.5F fenestrated and cuffed pleural and peritoneal catheters on either end (Fig 1). The size of these catheters has proved suitable for use in patients with a wide range of body weights, from infancy to adolescence. The shunt is implanted with a minimally invasive surgical procedure and allows for chyle to be pumped into the peritoneal cavity where it is reabsorbed without the loss of nutrients, lymphocytes, and electrolytes associated with tube thoracostomy.
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Material and methods
We reviewed the records of 15 patients treated with pleuro- or pericardialperitoneal shunts (Denver Biomaterials) at The Children’s Medical Center at the University of Virginia and at Cardinal Glennon Children’s Hospital at Saint Louis University Health Sciences Center between 1988 and 1998. Upon recognition of chylothorax or chylopericardium, conservative therapy was undertaken for variable periods of time. Therapy consisted of drainage of the pleural space by thoracentesis or tube thoracostomy, and restriction of oral fat intake to medium-chain triglycerides or the use of total parenteral nutrition. If a patient’s condition failed to improve or began to deteriorate, a pleuroperitoneal or pericardialperitoneal shunt was placed. Primary diagnosis, age at shunt placement, location of shunt, duration of chylothorax or chylopericardium prior to and following shunt placement, complications, and overall outcome of shunt treatment are shown in Table 1.
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Immediately after placement, the shunt was pumped repeatedly until the chamber no longer filled, indicating that the hemithorax was cleared of fluid. The patients were placed on a daily schedule of pumping based on the volume of fluid produced per day and the parents were instructed on how to use the pump. Usually this involved pumping the chamber every 2 to 4 hours until the chest was empty. Pumping frequency was reduced as the amount of chylous effusion declined, although the parents were instructed to pump the chamber at least twice daily to ensure shunt patency. Shunt therapy was considered successful and elective removal of the shunt was undertaken when serial radiographs showed resolution of the effusion over a 1-month period of time, during which the pump was not compressed.
Results
Fifteen patients were treated for a total of 19 chylous effusions (11 in the right pleural cavity, 6 in the left pleural cavity, and 2 in the pericardium). Patients ranged in age from 1 month to 11.5 years (mean: 2.1 years; median: 3 months). The most common indication (7 of 15) was refractory chylothorax following surgical correction of congenital heart disease. The duration of the chylothorax prior to shunt placement varied from 5 to 810 days with a mean duration of 76 days and a median duration of 14 days. Fourteen of the 17 pleural effusions (83%) resolved secondary to shunt therapy in an average of 95 days (range: 12 to 365 days). Both of the pericardial-peritoneal shunts were associated with successful outcomes. In all cases in which the chylous effusions cleared, the shunts were removed without further complications (Fig 2).
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During the course of the shunt therapy, there were a total of six shunt malfunctions that required intervention. Five of the six malfunctions involved shunt or valve obstruction and the other involved a broken pump valve. Shunt obstruction was managed by dividing the afferent limb and flushing the system with sterile saline. Some episodes of shunt obstruction due to omentum were managed by removing the peritoneal end of the shunt, removing the offending omentum from the shunt lumen, and replacing the shunt within the peritoneal cavity. Shunt obstruction resulted in system failure in 2 of 3 patients. Additionally, there were two shunt-associated infections that precipitated shunt removal. No infections resulted in failure of shunt therapy. Four male patients developed inguinal or umbilical hernia during therapy requiring repair.
Comment
Externalized pleuroperitoneal and pericardial-peritoneal shunts are safe and effective therapies for children with refractory chylous effusions. Pleuroperitoneal shunts alleviate the respiratory symptoms of chylothorax without the nutritional and immunological depletion or metabolic instability associated with long-term tube thoracostomy. This method of treatment allows pleural effusions to resolve without resorting to pleurodesis or thoracic duct ligation. Externalized shunt therapy also empowers parents who often feel helpless when their child is sick by involving them directly in their child’s healing process.
The pleuroperitoneal shunt with the exteriorized pump chamber has significant advantages over the original subcutaneous version of the shunt. Not only is placement of these shunts technically simpler, but exteriorization of the pump chamber allows for a large volume pump chamber, even in small infants. Because the pump chamber is larger and exteriorized, the pump needs to be compressed less frequently and the compressions are less painful and disturbing to the young patient. There are theoretical disadvantages to the externalized system, including infection and the need for frequent dressing changes.
All of the chylous effusions that failed to resolve during the course of shunt therapy were in patients with conditions associated with elevated vena cava or right atrial pressures. Although some researchers have reported success with pleuroperitoneal shunting after Fontan procedures [11, 12], there is substantial evidence to suggest that chylous effusions in patients with elevated right atrial or vena caval pressures, such as those that often exist after a Fontan procedure or with vena cava thrombosis, are more difficult to manage [10, 11]. In this subset of patients, the efficacy of pleuroperitoneal shunting requires further investigation.
We recommend externalized pleuroperitoneal shunts for the therapy of chylous effusions to avoid the problems associated with long-term chyle loss and respiratory compromise. This approach avoids a major operative procedure or pleurodesis, although these treatments may be necessary for patients in whom shunt therapy fails.
We propose a general treatment algorithm (Fig 3 ) that advocates pleuroperitoneal shunting of some refractory chylothoraces after 2 weeks of conservative therapy. Patients in whom a localized injury to the thoracic duct has occurred (eg, penetrating trauma) are likely to benefit from early thoracic duct ligation, rather than from shunt therapy. Shunt therapy is most appropriate for patients in whom the chylous leak is more diffuse (eg, postcardiotomy).
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