Interactive Cardiovascular and Thoracic Surgery 2:348-349(2003)
© 2003 European Association of Cardio-Thoracic Surgery
Successful medical treatment of persistent pleural drainage after the Fontan operation
Thomas Walthera,*,
Peter Theuneb,
Ian Sullivanb and
Marc R. de Levalb
a Klinik für Herzchirurgie, Herzzentrum, Universität Leipzig, Strümpellstrß 39, 04289 Leipzig, Germany
b Great Ormond Street Hospital for Children NHS Trust, London, UK
* Corresponding author. Tel.: +44-20-7573-8888; fax: +44-20-7573-8801
walt{at}medizin.uni-leipzig.de
Received March 17, 2003;
received in revised form April 30, 2003;
accepted May 2, 2003
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Abstract
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Prolonged pleural drainage remains the most serious complication after the Fontan operation with considerable morbidity and resource implication. Standard treatment including low fat diet, protein replacement, diuretics and angiotensin converting enzyme inhibitor therapy is not always effective. We report a patient in whom excessive and persistent drainage was finally treated with a somatostatin analogue successfully.
Key Words: Total cavopulmonary connection; Pleural effusion; Somatostatin
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1. Introduction
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Prolonged pleural drainage remains the most serious complication after the Fontan operation with considerable morbidity as well as resource implication [1]. At present, no completely effective therapy is available. Standard treatment includes low fat diet, protein replacement, diuretics and angiotensin converting enzyme (ACE) inhibitor therapy, all having variable success rates. Octreotide (SandostatinTM), an analogue of the hypothalamic release-inhibiting hormone somatostatin with antisecretory and antidiarrheal properties was reported to have beneficial effects in the treatment of chylothorax. We postulated that this effect could be exploited in the treatment of post-Fontan pleural drainage.
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2. Methods and results
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A 5.2-year-old patient with tricuspid atresia and severe pulmonary stenosis had received right modified Blalock–Taussig shunt at an age of 8 months followed by bidirectional cavopulmonary anastomosis with creation of pulmonary atresia and closure of the shunt at the age of 3.5 years. The first operation was complicated by temporary right phrenic palsy and the second one by right sided chylothorax. The patient was referred to us for completion of total cavopulmonary connection. On admission, he was 17 kg and moderately cyanosed with an oxygen saturation of 75%. Echocardiography revealed excellent left ventricular function, a good size atrial septal defect and only trivial mitral valve regurgitation. At cardiac catheter, mean pulmonary artery (PA) pressure was 7 mmHg with a widely patent cavopulmonary anastomosis.
Completion of the total cavopulmonary connection was performed using a 20 mm extracardiac Goretex graft. Hemodynamic function was satisfactory and he was extubated 4 h postoperatively with a central venous pressure of 10–12 mmHg. Inotropic support was quickly weaned and echocardiography revealed good ventricular and valve function. Postoperative drainage persisted being chylus from both pleural spaces and a low fat diet was commenced. Half of the drain losses were replaced with 4.5% albumin, and routine diuretics and ACE inhibitor therapy were started. When drainage persisted in the range of 600–1200 ml daily into the third postoperative week, repeat cardiac catheterisation was performed. There was unobstructed blood flow throughout, no evidence of intracardiac shunting, good ventricular function and a competent mitral valve. Pressures in the superior vena cava (SVC), inferior vena cava (IVC), right and left pulmonary arteries (RPA and LPA) were all similar at a mean of 11–12 mmHg. Multiple collateral arteries to the RPA were treated by coil occlusion of the distal right internal mammary artery. However, drainage further persisted, and total parenteral nutrition (TPN) was then started on POD 30. One week later, he developed sepsis, requiring reintubation for 5 days and antibiotic and antifungal therapy for several weeks. TPN was discontinued because it did not have any effect on drainage, which persisted in excess of 500 ml daily. It was then decided on POD 60 to start intravenous Octreotide at a dose of 3.5 µg/kg/h initially, then being increased to 7 µg/kg/h. Over the next 3.5 weeks, pleural drainage initially diminished to 100 ml/day, then peaked again to about 300 ml/day and finally, decreased to below 20 ml/day. The drains were removed on POD 90, Octreotide was discontinued, no side effects occurred, and the patient was discharged 5 days later. At 6 months follow-up, the patient was well without any recurrence.
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3. Discussion
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Prolonged postoperative pleural fluid losses are thought to be caused by the elevated systemic venous pressures associated with the Fontan circulation. However, in our patient, drainage occurred despite excellent hemodynamics. The exact pathophysiological pathways of prolonged pleural drainage after Fontan operation are not fully understood. Persisting drainage may be related to fluid that normally returns to the circulation via the lymphatic system. Chylus would probably come from the gut lymphatic system whereas serous fluid would be from the intrathoracic lymphatic system. No sufficiently effective therapeutic approach is available at present.
Octreotide is an effective antisecretory and antidiarrheal agent clinically used for the treatment of esophageal varices, diarrhea or small bowel fistulas for example. Its use has been reported in some individual patients with chylothorax. These reports include a 4-month-old child after a Senning procedure [2], an adult after coronary artery bypass grafting [3], two patients with postsurgical lymphatic leak [4] and one with spontaneous chylothorax [5]. Furthermore, experimental studies have been performed revealing the effectiveness of Octreotide medication in an animal model after thoracic duct transsection [6]. Although the exact mechanism of action of Octreotide remains to be clarified, it had been postulated that it could be beneficial to decrease post-Fontan pleural drainage. In our patient, once Octreotide was started on the 60th POD, the pleural drainage decreased subsequently within a few days and subsided after 4 weeks. However, the association may be coincidental rather than causal. No side effects occurred, nevertheless, the circulatory, endocrine, gastrointestinal and liver function should be carefully monitored during treatment.
Octreotide was only started after persisting drainage and after no other interventions proved to be effective. However, immediate treatment could be beneficial to decrease drainage and improve outcome after the Fontan operation. If further effects can be proven, this medication may reach more widespread application. To prove the effects of Octreotide for post-Fontan effusions, a double blind randomized multicenter evaluation should be initiated.
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Appendix A.
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ICVTS on-line discussion
Author: Enrico Aidala, Osp. Infantile "Regina Margherita", Pediatric Cardiac Surgery, P.zza Polonia 94, Torino, 10126 Italy
Date: 09-Jul-03 16:26
Message: The use of octreotide or, previously, somatostatin for chylothorax after cardiac surgery, particularly Fontan-type operation, has been suggested in recent years (i.e. Ann Thorac Surg 2001;72:1740-2). We used it in five cases of Fontan procedures in the last 2 years, with good results, on average in 10 days. The main side effects of its use are nausea, vomiting, melena and abdominal pain; we experienced them once and we used e.v. infusion up to 4 mcg/Kg/h. The authors used octreotide up to 7 mcg/Kg/h for about 25 days, without side effects. Do they have knowledge of the maximal safe dosage and duration of this therapy?
doi:10.1016/S1569-9293(03)00103-8
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References
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Abstract
1. Introduction