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Octreotide therapy in two children with intractable postoperative chylothorax
International Journal of Cardiology 146 (2011) e63 – e65 www.elsevier.com/locate/ijcard
Letter to the Editor
Octreotide therapy in two children with intractable postoperative chylothorax Wen-Pin Hung a , Jieh-Neng Wang a , Hok-Keong Chang b , Jing-Ming Wu a,⁎ a
Department of Pediatrics Cardiology, National Cheng Kung University Hospital, No.138 Sheng-Li Road, Tainan 70403, Taiwan b Department of Pediatrics, Sin-Lau Hospital, Madou Branch, Tainan, Taiwan Received 26 December 2008; accepted 29 December 2008 Available online 3 February 2009
Abstract Postoperative chylothorax is sometimes difficult to be managed either by conventional therapies or by surgery. Herein, we report the experience of octreotide therapy for intractable postoperative chylothorax in two children (one was severe tetralogy of Fallot received Blalock–Taussing shunt, and the other was a single ventricle received bi-directional Glenn shunt) and review the literature on octreotide efficacy. © 2009 Elsevier Ireland Ltd. All rights reserved. Keywords: Chylothorax; Postoperative; Octreotide; Somatostatin; Congenital heart disease
1. Case report 1.1. Case 1 A 5 day-old female newborn, presented with marked cyanosis and dyspnea, was diagnosed as severe tetralogy of Fallot with tiny patent ductus arteriosus. Despite of prostaglandin E1 infusion the symptoms persisted, so she received creation of a left-side modified Blalock–Taussing shunt on the 7th day of her life. After the operation, unexpected plenty amount of pleural effusion (about 80 ml/Kg/day) was noted in the first few days. Chylothorax was highly suspected, however, the triglyceride (TG) concentration in pleural effusion was not elevated (38 mg/dl) at that time. Then we delayed oral feeding and supported nutrition by TPN since postoperative day 4 (POD 4). After the above management, the amount of pleural effusion mildly decreased in the following days so we resumed oral feeding with regular-formula milk since POD 7. Un-
⁎ Corresponding author. Tel.: +886 6 2353535x4187; fax: +886 6 2753083. E-mail address: [email protected] (J.M. Wu). 0167-5273/$ - see front matter © 2009 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijcard.2008.12.210
fortunately, increasing amount of pleural effusion with whitish cloudy content was found after oral feeding. We rechecked the pleural effusion which then demonstrated elevated TG concentration (192 mg/dL) on POD 8. Chylothorax was definitely diagnosed. Due to persistent much amount of chlyothorax under conventional therapy and to prevent the possible lifethreatening complication of chylothorax, octreotide in the dose of 1 ug/kg/hr was administered since POD 8. The amount of pleural effusion decreased significantly thereafter (Fig. 1), and the pleural effusion turned to normal
Fig. 1. The drainage amount of chylothorax in case 1 significantly decreased after octreotide treatment.
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light-yellowish color after 2 days of octreotide treatment. Medium-chain-triglyceride (MCT)-rich milk formula was given since POD 10 after consulting the dietician. Octreotide was discontinued on POD 12 after a total 4 day treatment. Chest tube was removed on POD 14 since the amount of pleural effusion decreased to less than 10 mL/day. We resumed regular-formula milk 1 month later and no more recurrence of chylothorax was noted. 2. Case 2 A 19 month-old girl diagnosed as complex cyanotic congenital heart disease (double-inlet of single ventricle, transposition of the great arteries and pulmonary stenosis) at 8 days old. She received creation of bidirectional Glenn shunt (cavopulmonary connection) at 14 months old smoothly. However, mild pleural effusion was noted after operation but the amount did not increased obviously during follow-up at outpatient clinic. Unfortunately, she suffered from progressive shortness of breath and facial swelling 4 months after operation. The echocardiography demonstrated much amount of rightside pleural effusion. We then performed diagnostic chest tapping and the pleural effusion looked milk-like color. The analysis of pleural effusion revealed leukocytosis with lymphocyte predominant (98%) and elevated TG level (934 mg/dl). Chylothorax possibly related to high central venous pressure after cavopulmonary connection was diagnosed. We then inserted pig-tail for drainage and gave MCT-rich formula for feeding. After the above management, her pleural effusion decreased gradually and she was discharged 1 week later. However, shortness of breath with facial swelling attacked again 20 days later after discharge from hospital. The echocardiography showed much amount of right-side pleural effusion again and the analysis of pleural effusion confirmed the diagnosis of chylothorax. Due to recurrent episodes of chylothorax under conventional treatment, octreotide in the dose of 1 ug/kg/hr was administered. The amount of pleural effusion decreased significantly thereafter (Fig. 2), and the pleural effusion turned to normal light-yellowish color after 2 days of octreotde treatment. Octreotide was discontinued after a total 5 day treatment. We
also resumed regular diet 1 month later and no more episode of chylothorax attacked since then. 3. Discussion Chylothorax in children is rare but potentially lifethreatening. It might be congenital cause or a complication of different conditions including malignancy, surgery and trauma [1]. Post-cardiothoracic surgery is one of common causes of secondary chylothorax. The mechanisms of postoperative chylothorax were most due to direct trauma to the lymphatic vessels or the thoracic duct (like case 1), and less common secondary to central venous hypertension after cavopulmonary connection (like case 2), or venous thrombosis [2]. The anatomic location of thoracic duct makes it vulnerable during intrathoracic surgeries. The injured level of thoracic duct, above or below thoracic vertebra 5 and 6, may decide the occurrence of left-side (like case 1) or right-side (like case 2) chylothorax, respectively. In addition to pleural catheter insertion, the first-line treatment for chylothorax conventionally consists of enteric rest with total parenteral nutrition (TPN) or medium-chaintriglyceride (MCT) diet, which may require months of conservative intensive care treatment [3]. Surgical ligation of the thoracic duct is recommended for intractable chylothorax, while it is associated with a high failure rate. Sometimes postoperative chylothorax is difficult to be managed either by conventional therapies or by surgery. Octreotide, a synthetic SST analogue with the advantage of a much longer half-life (2–6 h) than SST, has been employed for similar situation as SST and recently also for treating chylothorax in pediatric patients [4]. Acclaimed positive attributes of octreotide therapy include a shorter duration of intensive care treatment, a reduction of recurrent thoracocentesis, and fewer fluid and plasma infusions, thereby reducing the risk of infection [5]. However, the mechanism by which octreotide decreases chyle production remains unknown. The reported side effects of octreotide in children included transient changes in blood glucose levels, transient abdominal distension and emesis. In our two patients, intravenous octreotide infusion reduced the chyle production significantly 2 days later when used at a dose of 1 ug/kg/hr. The use of octreotide in our patients was safe and there were no obvious side effects. In conclusion, octreotide might be an effectively therapeutic alternative in patient with prolonged postoperative chylothorax. Acknowledgments The author of this manuscript has certified that he complies with the Principles of Ethical Publishing in the International Journal of Cardiology [6]. References
Fig. 2. The drainage amount of chylothorax in case 2 significantly decreased after octreotide treatment.
[1] Beghetti M, La Scala G, Belli D, Bugmann P, Kalangos A, Le Coultre C. Etiology and management of pediatric chylothorax. J Pediatr 2000;136: 653–8.
W.P. Hung et al. / International Journal of Cardiology 146 (2011) e63–e65 [2] Efrati O, Barak A. Pleural effusions in the pediatric population. Pediatr Rev 2002;12:417–26. [3] Buttiker V, Fanconi S, Burger R. Chylothorax in children: guidelines for diagnosis and management. Chest 1999;116:682–7. [4] Hamdan MA, Gaeta ML. Octreotide and low-fat breast milk in postoperative chylothorax. Ann Thorac Surg 2004;77:2215–7.
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[5] Rosti L, De Battisti F, Butera G, Cirri S, Chessa M, Delogu A, et al. Octreotide in the management of postoperative chylothrax. Pediatr Cardiol 2005;26:440–3. [6] Coats AJ. Ethical authorship and publishing. Int J Cardiol 2009;131: 149–50.
Letter to the Editor
Octreotide therapy in two children with intractable postoperative chylothorax Wen-Pin Hung a , Jieh-Neng Wang a , Hok-Keong Chang b , Jing-Ming Wu a,⁎ a
Department of Pediatrics Cardiology, National Cheng Kung University Hospital, No.138 Sheng-Li Road, Tainan 70403, Taiwan b Department of Pediatrics, Sin-Lau Hospital, Madou Branch, Tainan, Taiwan Received 26 December 2008; accepted 29 December 2008 Available online 3 February 2009
Abstract Postoperative chylothorax is sometimes difficult to be managed either by conventional therapies or by surgery. Herein, we report the experience of octreotide therapy for intractable postoperative chylothorax in two children (one was severe tetralogy of Fallot received Blalock–Taussing shunt, and the other was a single ventricle received bi-directional Glenn shunt) and review the literature on octreotide efficacy. © 2009 Elsevier Ireland Ltd. All rights reserved. Keywords: Chylothorax; Postoperative; Octreotide; Somatostatin; Congenital heart disease
1. Case report 1.1. Case 1 A 5 day-old female newborn, presented with marked cyanosis and dyspnea, was diagnosed as severe tetralogy of Fallot with tiny patent ductus arteriosus. Despite of prostaglandin E1 infusion the symptoms persisted, so she received creation of a left-side modified Blalock–Taussing shunt on the 7th day of her life. After the operation, unexpected plenty amount of pleural effusion (about 80 ml/Kg/day) was noted in the first few days. Chylothorax was highly suspected, however, the triglyceride (TG) concentration in pleural effusion was not elevated (38 mg/dl) at that time. Then we delayed oral feeding and supported nutrition by TPN since postoperative day 4 (POD 4). After the above management, the amount of pleural effusion mildly decreased in the following days so we resumed oral feeding with regular-formula milk since POD 7. Un-
⁎ Corresponding author. Tel.: +886 6 2353535x4187; fax: +886 6 2753083. E-mail address: [email protected] (J.M. Wu). 0167-5273/$ - see front matter © 2009 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijcard.2008.12.210
fortunately, increasing amount of pleural effusion with whitish cloudy content was found after oral feeding. We rechecked the pleural effusion which then demonstrated elevated TG concentration (192 mg/dL) on POD 8. Chylothorax was definitely diagnosed. Due to persistent much amount of chlyothorax under conventional therapy and to prevent the possible lifethreatening complication of chylothorax, octreotide in the dose of 1 ug/kg/hr was administered since POD 8. The amount of pleural effusion decreased significantly thereafter (Fig. 1), and the pleural effusion turned to normal
Fig. 1. The drainage amount of chylothorax in case 1 significantly decreased after octreotide treatment.
e64
W.P. Hung et al. / International Journal of Cardiology 146 (2011) e63–e65
light-yellowish color after 2 days of octreotide treatment. Medium-chain-triglyceride (MCT)-rich milk formula was given since POD 10 after consulting the dietician. Octreotide was discontinued on POD 12 after a total 4 day treatment. Chest tube was removed on POD 14 since the amount of pleural effusion decreased to less than 10 mL/day. We resumed regular-formula milk 1 month later and no more recurrence of chylothorax was noted. 2. Case 2 A 19 month-old girl diagnosed as complex cyanotic congenital heart disease (double-inlet of single ventricle, transposition of the great arteries and pulmonary stenosis) at 8 days old. She received creation of bidirectional Glenn shunt (cavopulmonary connection) at 14 months old smoothly. However, mild pleural effusion was noted after operation but the amount did not increased obviously during follow-up at outpatient clinic. Unfortunately, she suffered from progressive shortness of breath and facial swelling 4 months after operation. The echocardiography demonstrated much amount of rightside pleural effusion. We then performed diagnostic chest tapping and the pleural effusion looked milk-like color. The analysis of pleural effusion revealed leukocytosis with lymphocyte predominant (98%) and elevated TG level (934 mg/dl). Chylothorax possibly related to high central venous pressure after cavopulmonary connection was diagnosed. We then inserted pig-tail for drainage and gave MCT-rich formula for feeding. After the above management, her pleural effusion decreased gradually and she was discharged 1 week later. However, shortness of breath with facial swelling attacked again 20 days later after discharge from hospital. The echocardiography showed much amount of right-side pleural effusion again and the analysis of pleural effusion confirmed the diagnosis of chylothorax. Due to recurrent episodes of chylothorax under conventional treatment, octreotide in the dose of 1 ug/kg/hr was administered. The amount of pleural effusion decreased significantly thereafter (Fig. 2), and the pleural effusion turned to normal light-yellowish color after 2 days of octreotde treatment. Octreotide was discontinued after a total 5 day treatment. We
also resumed regular diet 1 month later and no more episode of chylothorax attacked since then. 3. Discussion Chylothorax in children is rare but potentially lifethreatening. It might be congenital cause or a complication of different conditions including malignancy, surgery and trauma [1]. Post-cardiothoracic surgery is one of common causes of secondary chylothorax. The mechanisms of postoperative chylothorax were most due to direct trauma to the lymphatic vessels or the thoracic duct (like case 1), and less common secondary to central venous hypertension after cavopulmonary connection (like case 2), or venous thrombosis [2]. The anatomic location of thoracic duct makes it vulnerable during intrathoracic surgeries. The injured level of thoracic duct, above or below thoracic vertebra 5 and 6, may decide the occurrence of left-side (like case 1) or right-side (like case 2) chylothorax, respectively. In addition to pleural catheter insertion, the first-line treatment for chylothorax conventionally consists of enteric rest with total parenteral nutrition (TPN) or medium-chaintriglyceride (MCT) diet, which may require months of conservative intensive care treatment [3]. Surgical ligation of the thoracic duct is recommended for intractable chylothorax, while it is associated with a high failure rate. Sometimes postoperative chylothorax is difficult to be managed either by conventional therapies or by surgery. Octreotide, a synthetic SST analogue with the advantage of a much longer half-life (2–6 h) than SST, has been employed for similar situation as SST and recently also for treating chylothorax in pediatric patients [4]. Acclaimed positive attributes of octreotide therapy include a shorter duration of intensive care treatment, a reduction of recurrent thoracocentesis, and fewer fluid and plasma infusions, thereby reducing the risk of infection [5]. However, the mechanism by which octreotide decreases chyle production remains unknown. The reported side effects of octreotide in children included transient changes in blood glucose levels, transient abdominal distension and emesis. In our two patients, intravenous octreotide infusion reduced the chyle production significantly 2 days later when used at a dose of 1 ug/kg/hr. The use of octreotide in our patients was safe and there were no obvious side effects. In conclusion, octreotide might be an effectively therapeutic alternative in patient with prolonged postoperative chylothorax. Acknowledgments The author of this manuscript has certified that he complies with the Principles of Ethical Publishing in the International Journal of Cardiology [6]. References
Fig. 2. The drainage amount of chylothorax in case 2 significantly decreased after octreotide treatment.
[1] Beghetti M, La Scala G, Belli D, Bugmann P, Kalangos A, Le Coultre C. Etiology and management of pediatric chylothorax. J Pediatr 2000;136: 653–8.
W.P. Hung et al. / International Journal of Cardiology 146 (2011) e63–e65 [2] Efrati O, Barak A. Pleural effusions in the pediatric population. Pediatr Rev 2002;12:417–26. [3] Buttiker V, Fanconi S, Burger R. Chylothorax in children: guidelines for diagnosis and management. Chest 1999;116:682–7. [4] Hamdan MA, Gaeta ML. Octreotide and low-fat breast milk in postoperative chylothorax. Ann Thorac Surg 2004;77:2215–7.
e65
[5] Rosti L, De Battisti F, Butera G, Cirri S, Chessa M, Delogu A, et al. Octreotide in the management of postoperative chylothrax. Pediatr Cardiol 2005;26:440–3. [6] Coats AJ. Ethical authorship and publishing. Int J Cardiol 2009;131: 149–50.