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Chylous ascites after excision of a choledochal cyst in a child
Journal of Pediatric Surgery (2009) 44, E5–E7
www.elsevier.com/locate/jpedsurg
Chylous ascites after excision of a choledochal cyst in a child Chun-Chieh Huang, Ming-Shian Tsai, Hong-Shiee Lai ⁎ Department of Surgery, National Taiwan University Hospital and National Taiwan University College of Medicine, Taipei, Taiwan Received 26 November 2008; revised 27 January 2009; accepted 28 January 2009
Key words: Chylous ascites; Choledochal cyst; Somatostatin
Abstract We report a case of chylous ascites developing 2 weeks after excision of a choledochal cyst with a Roux-en-Y hepaticojejunostomy. Despite the failure of the initial attempts to resolve the chylous ascites by fasting, subsequently, we successfully treated the patient's chylous ascites with intravenous somatostatin. No obvious adverse side effects occurred during the use of somatostatin. To the best of our knowledge, this is the first report of chylous ascites after choledochal cyst excision in children, which was successfully treated with somatostatin. Somatostatin may be considered as a therapeutic option of managing pediatric postoperative chylous ascites. Crown Copyright © 2009 Published by Elsevier Inc. All rights reserved.
Chylous ascites is the extravasation of milky chyle rich in triglycerides into the peritoneal cavity. It develops when the lymphatic system becomes obstructed or disrupted. The most common causes are abdominal malignancy in adults and congenital lymphatic abnormalities in children [1]. Postoperative chylous ascites is not so rare in the pediatric population, but no case has been reported in the English literature after excision of a choledochal cyst in children. Only one case has been reported after hepaticojejunostomy for spontaneous biliary perforation [2]. Herein, we reported a case of chylous ascites that occurred after excision of a choledochal cyst with a Roux-en-Y hepaticojejunostomy that was treated successfully with fasting and intravenous somatostatin.
⁎ Corresponding author. Department of Surgery, National Taiwan University Hospital, Taipei, Taiwan 100, R.O.C. Tel.: +886 2 23123456x65112, 65116; fax: +886 2 23223870. E-mail address: [email protected] (H.-S. Lai).
1. Case report A 2-year 9-month-old boy had a type I choledochal cyst (Fig. 1) and had experienced repeated abdominal pain and progressive jaundice for 4 months. At the time of his operation, we found a thickened gallbladder with moderate adhesions around the hepatic hilum. The dilated common bile duct and common hepatic duct was 17 mm in diameter. We also observed one aberrant hepatic duct extending from the right liver lobe draining directly into the choledochal cyst. After cholecystectomy and total excision of the choledochal cyst, we performed a Roux-en-Y hepaticojejunostomy with 2 anastomoses of the jejunal stump to the common hepatic duct and to the aberrant hepatic duct. The surgical procedures went smoothly, and a Penrose drain was placed in the right upper quadrant, near the anastomoses. After the procedure, the child was kept without oral intake and received total parenteral nutrition (TPN). On postoperative day (POD) 7, the amount of drainage fluid from the Penrose drain increased to 144 mL, and the
0022-3468/$ – see front matter. Crown Copyright © 2009 Published by Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2009.01.076
E6
C.-C. Huang et al. PTE Ltd) from POD 15. The dose of somatostatin was 6 μg/ kg per hour given intravenously. The amounts of chylous drainage, decreased dramatically after the use of somatostatin from POD 15 to POD 20, were 220 mL, 96 mL, 133 mL, 45 mL, 22 mL, and 8 mL, respectively (Fig. 3). Oral intake was resumed on POD 21 and did not induce recurrence of the chylous ascites. The Penrose drain was removed on POD 23. The child recovered well and was discharged on POD 26. Abdominal sonography 2 months later showed no recurrence of chylous ascites, and he was tolerating a normal diet.
2. Discussion
Fig. 1 Magnetic resonance cholangiography showed cylindrical dilatation of the common bile duct, hepatic ducts, and cystic duct.
contents were greenish and whitish. The levels of total bilirubin, amylase, and lipase in the drainage fluid were 0.62 mg/dL, 45 U/L, and 40 U/L, respectively. The nasogastric tube was removed on POD 9, but the Penrose drainage became slightly turbid on POD 11. Suspecting leakage from an anastomotic site, we performed an upper gastrointestinal series with oral contrast medium (urograffin) and methylene blue on POD 12. Serial plain abdominal radiographs revealed no specific leakage of contrast, and there was also no methylene blue staining from the Penrose drain. Enteral feeding was started on POD 13. However, 202 mL milky turbid drainage from the Penrose drain tube was noted on POD 14 (Fig. 2). Biochemical analysis revealed a triglyceride content of 2180 mg/dL and a cholesterol content of 54 mg/dL. We diagnosed chylous ascites and prescribed TPN for bowel rest and somatostatin (Stilamin, Serono Singapore
Fig. 2 The turbid drainage from the Penrose drain tube proved to be chylous ascites.
Postoperative chylous ascites is not so rare in the pediatric population. They more commonly occur after retroperitoneal operations such as nephrectomy, retroperitoneal lymph node dissection for nephroblastoma, and testis cancer [3]. Other cases have been reported after repair of congenital diaphragmatic hernia [4], malrotation and gastroschisis [5,6], Nissen fundoplication [7], duodenotomy [8], liver resection [9], liver transplantation [10], and hepaticojejunostomy [2]. The reported complications after excision of choledochal cysts include cholangitis, intrahepatic gallstones, biliary strictures, and bowel obstruction [11-13]; however, chylous ascites has not yet been reported. The site of chyle leakage in children may not be easily recognized. This is the only instance of chylous ascites in our experience with 107 choledochal cysts in children in the past 18 years. Because an aberrant hepatic duct was noted in this unique case, an anomaly of the lymphatic duct drainage routes may have also been present. During the surgical procedure, an unrecognized injury to the abnormal lymphatics in the root of mesentery or hepatic
Fig. 3 Time course of the postoperative drainage amount. Note that the chylous ascites decreased immediately and dramatically after starting somatostatin in this patient.
Chylous ascites and choledochal cyst hilum may have occurred. Compression or injury to other congested abdominal lymph vessels by manipulation could also be considered. The treatment of chylous ascites is primarily conservative and is intended to decrease the lymph flow to a leak and replenish the nutritional losses. In 2002, Leibovitch et al [3] proposed an algorithm for treating postoperative chylous ascites with a step-by-step management plan including repeat paracentesis, low-fat diet with medium-chain triglycerides, fasting with TPN, administration of somatostatin, and surgery or peritoneovenous shunting reserved for refractory cases. However, this algorithm was based on literature focusing mostly on adult patients. Few reports have been published on the feasibility of managing chylous ascites in pediatric patients. Given this limited experience with postoperative chylous ascites in children, the optimal management of such patients is not well defined. Somatostatin is known to reduce splanchnic, hepatic, and portal blood flow and to inhibit intestinal motility and secretion; thus, it can reduce lymph flow. Because somatostatin interferes with blood glucose regulation, close monitoring of blood glucose and gradual tapering of somatostatin is recommended. Satisfactory results have been reported in adults, but in those cases, somatostatin was used as a second-line treatment [3]. A somatostatin analog was first used for postoperative chylous ascites in a pediatric patient in 2001, as reported by Bhatia et al [8]. A review of the literature shows only 4 pediatric case reports of postoperative chylous ascites that were successfully treated with a somatostatin analog [4,8,10]. In those reports, the treatment course was within 1 to 2 weeks without side effects. In our patient, chylous ascites occurred although the patient was fasting for 2 weeks and increased after oral intake but resolved immediately after the use of somatostatin (Fig. 3). This provided indirect evidence that somatostatin may treat chylous ascites effectively, though the causality still needs further investigation. Moreover, with cautious use of somatostatin for a limited period of 5 days, we found no severe side effects occurring in this patient. The patient was discharged 11 days after the use of somatostatin.
E7 To the best of our knowledge, this is the first report of chylous ascites after choledochal cyst excision in children, which was successfully treated with somatostatin. Chylous ascites should be kept in mind as a possible complication of choledochal cyst excision. On the basis of previous reports and our experience, somatostatin should be considered as a possible therapeutic option in managing pediatric postoperative chylous ascites.
References [1] Aalami OO, Allen DB, Organ Jr CH. Chylous ascites: a collective review. Surgery 2000;128:761-78. [2] Livesey E, Davenport M. Spontaneous perforation of the biliary tract and portal vein thrombosis in infancy. Pediatr Surg Int 2008;24:357-9. [3] Leibovitch I, Mor Y, Golomb J, et al. The diagnosis and management of postoperative chylous ascites. J Urol 2002;167:449-57. [4] Andreou A, Papouli M, Papavasiliou V, et al. Postoperative chylous ascites in a neonate treated successfully with octreotide: bile sludge and cholestasis. Am J Perinatol 2005;22:401-4. [5] Lloyd DA. Gastroschisis, malrotation, and chylous ascites. J Pediatr Surg 1991;26:106-7. [6] Drinnen D, Filston HC. An unusual case of gastroschisis with gallbladder evisceration and late postrepair chylous ascites. J Pediatr Surg 1997;32:1804-5. [7] Parys SC, Hart MH. Chylous ascites following a Nissen fundoplication. J Pediatr Gastroenterol Nutr 1992;15:181-3. [8] Bhatia C, Pratap U, Slavik Z. Octreotide therapy: a new horizon in treatment of iatrogenic chyloperitoneum. Arch Dis Child 2001;85: 234-5. [9] Prabhakaran K, Vidyadhar M, Jahoorahmad P, et al. Chylous ascites following liver resection—case report. Pediatr Surg Int 2004;20: 719-21. [10] Baran M, Cakir M, Yüksekkaya HA, et al. Chylous ascites after living related liver transplantation treated with somatostatin analog and parenteral nutrition. Transplant Proc 2008;40:320-1. [11] Todani T, Watanabe Y, Urushihara N, et al. Biliary complications after excisional procedure for choledochal cyst. J Pediatr Surg 1995;30: 478-81. [12] Yamataka A, Ohshiro K, Okada Y, et al. Complications after cyst excision with hepaticoenterostomy for choledochal cysts and their surgical management in children versus adults. J Pediatr Surg 1997;32: 1097-102. [13] Yamataka A, Ohshiro K, Okada Y, et al. Early and late results of excision of choledochal cysts. J Pediatr Surg 1997;32:1563-6.
www.elsevier.com/locate/jpedsurg
Chylous ascites after excision of a choledochal cyst in a child Chun-Chieh Huang, Ming-Shian Tsai, Hong-Shiee Lai ⁎ Department of Surgery, National Taiwan University Hospital and National Taiwan University College of Medicine, Taipei, Taiwan Received 26 November 2008; revised 27 January 2009; accepted 28 January 2009
Key words: Chylous ascites; Choledochal cyst; Somatostatin
Abstract We report a case of chylous ascites developing 2 weeks after excision of a choledochal cyst with a Roux-en-Y hepaticojejunostomy. Despite the failure of the initial attempts to resolve the chylous ascites by fasting, subsequently, we successfully treated the patient's chylous ascites with intravenous somatostatin. No obvious adverse side effects occurred during the use of somatostatin. To the best of our knowledge, this is the first report of chylous ascites after choledochal cyst excision in children, which was successfully treated with somatostatin. Somatostatin may be considered as a therapeutic option of managing pediatric postoperative chylous ascites. Crown Copyright © 2009 Published by Elsevier Inc. All rights reserved.
Chylous ascites is the extravasation of milky chyle rich in triglycerides into the peritoneal cavity. It develops when the lymphatic system becomes obstructed or disrupted. The most common causes are abdominal malignancy in adults and congenital lymphatic abnormalities in children [1]. Postoperative chylous ascites is not so rare in the pediatric population, but no case has been reported in the English literature after excision of a choledochal cyst in children. Only one case has been reported after hepaticojejunostomy for spontaneous biliary perforation [2]. Herein, we reported a case of chylous ascites that occurred after excision of a choledochal cyst with a Roux-en-Y hepaticojejunostomy that was treated successfully with fasting and intravenous somatostatin.
⁎ Corresponding author. Department of Surgery, National Taiwan University Hospital, Taipei, Taiwan 100, R.O.C. Tel.: +886 2 23123456x65112, 65116; fax: +886 2 23223870. E-mail address: [email protected] (H.-S. Lai).
1. Case report A 2-year 9-month-old boy had a type I choledochal cyst (Fig. 1) and had experienced repeated abdominal pain and progressive jaundice for 4 months. At the time of his operation, we found a thickened gallbladder with moderate adhesions around the hepatic hilum. The dilated common bile duct and common hepatic duct was 17 mm in diameter. We also observed one aberrant hepatic duct extending from the right liver lobe draining directly into the choledochal cyst. After cholecystectomy and total excision of the choledochal cyst, we performed a Roux-en-Y hepaticojejunostomy with 2 anastomoses of the jejunal stump to the common hepatic duct and to the aberrant hepatic duct. The surgical procedures went smoothly, and a Penrose drain was placed in the right upper quadrant, near the anastomoses. After the procedure, the child was kept without oral intake and received total parenteral nutrition (TPN). On postoperative day (POD) 7, the amount of drainage fluid from the Penrose drain increased to 144 mL, and the
0022-3468/$ – see front matter. Crown Copyright © 2009 Published by Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2009.01.076
E6
C.-C. Huang et al. PTE Ltd) from POD 15. The dose of somatostatin was 6 μg/ kg per hour given intravenously. The amounts of chylous drainage, decreased dramatically after the use of somatostatin from POD 15 to POD 20, were 220 mL, 96 mL, 133 mL, 45 mL, 22 mL, and 8 mL, respectively (Fig. 3). Oral intake was resumed on POD 21 and did not induce recurrence of the chylous ascites. The Penrose drain was removed on POD 23. The child recovered well and was discharged on POD 26. Abdominal sonography 2 months later showed no recurrence of chylous ascites, and he was tolerating a normal diet.
2. Discussion
Fig. 1 Magnetic resonance cholangiography showed cylindrical dilatation of the common bile duct, hepatic ducts, and cystic duct.
contents were greenish and whitish. The levels of total bilirubin, amylase, and lipase in the drainage fluid were 0.62 mg/dL, 45 U/L, and 40 U/L, respectively. The nasogastric tube was removed on POD 9, but the Penrose drainage became slightly turbid on POD 11. Suspecting leakage from an anastomotic site, we performed an upper gastrointestinal series with oral contrast medium (urograffin) and methylene blue on POD 12. Serial plain abdominal radiographs revealed no specific leakage of contrast, and there was also no methylene blue staining from the Penrose drain. Enteral feeding was started on POD 13. However, 202 mL milky turbid drainage from the Penrose drain tube was noted on POD 14 (Fig. 2). Biochemical analysis revealed a triglyceride content of 2180 mg/dL and a cholesterol content of 54 mg/dL. We diagnosed chylous ascites and prescribed TPN for bowel rest and somatostatin (Stilamin, Serono Singapore
Fig. 2 The turbid drainage from the Penrose drain tube proved to be chylous ascites.
Postoperative chylous ascites is not so rare in the pediatric population. They more commonly occur after retroperitoneal operations such as nephrectomy, retroperitoneal lymph node dissection for nephroblastoma, and testis cancer [3]. Other cases have been reported after repair of congenital diaphragmatic hernia [4], malrotation and gastroschisis [5,6], Nissen fundoplication [7], duodenotomy [8], liver resection [9], liver transplantation [10], and hepaticojejunostomy [2]. The reported complications after excision of choledochal cysts include cholangitis, intrahepatic gallstones, biliary strictures, and bowel obstruction [11-13]; however, chylous ascites has not yet been reported. The site of chyle leakage in children may not be easily recognized. This is the only instance of chylous ascites in our experience with 107 choledochal cysts in children in the past 18 years. Because an aberrant hepatic duct was noted in this unique case, an anomaly of the lymphatic duct drainage routes may have also been present. During the surgical procedure, an unrecognized injury to the abnormal lymphatics in the root of mesentery or hepatic
Fig. 3 Time course of the postoperative drainage amount. Note that the chylous ascites decreased immediately and dramatically after starting somatostatin in this patient.
Chylous ascites and choledochal cyst hilum may have occurred. Compression or injury to other congested abdominal lymph vessels by manipulation could also be considered. The treatment of chylous ascites is primarily conservative and is intended to decrease the lymph flow to a leak and replenish the nutritional losses. In 2002, Leibovitch et al [3] proposed an algorithm for treating postoperative chylous ascites with a step-by-step management plan including repeat paracentesis, low-fat diet with medium-chain triglycerides, fasting with TPN, administration of somatostatin, and surgery or peritoneovenous shunting reserved for refractory cases. However, this algorithm was based on literature focusing mostly on adult patients. Few reports have been published on the feasibility of managing chylous ascites in pediatric patients. Given this limited experience with postoperative chylous ascites in children, the optimal management of such patients is not well defined. Somatostatin is known to reduce splanchnic, hepatic, and portal blood flow and to inhibit intestinal motility and secretion; thus, it can reduce lymph flow. Because somatostatin interferes with blood glucose regulation, close monitoring of blood glucose and gradual tapering of somatostatin is recommended. Satisfactory results have been reported in adults, but in those cases, somatostatin was used as a second-line treatment [3]. A somatostatin analog was first used for postoperative chylous ascites in a pediatric patient in 2001, as reported by Bhatia et al [8]. A review of the literature shows only 4 pediatric case reports of postoperative chylous ascites that were successfully treated with a somatostatin analog [4,8,10]. In those reports, the treatment course was within 1 to 2 weeks without side effects. In our patient, chylous ascites occurred although the patient was fasting for 2 weeks and increased after oral intake but resolved immediately after the use of somatostatin (Fig. 3). This provided indirect evidence that somatostatin may treat chylous ascites effectively, though the causality still needs further investigation. Moreover, with cautious use of somatostatin for a limited period of 5 days, we found no severe side effects occurring in this patient. The patient was discharged 11 days after the use of somatostatin.
E7 To the best of our knowledge, this is the first report of chylous ascites after choledochal cyst excision in children, which was successfully treated with somatostatin. Chylous ascites should be kept in mind as a possible complication of choledochal cyst excision. On the basis of previous reports and our experience, somatostatin should be considered as a possible therapeutic option in managing pediatric postoperative chylous ascites.
References [1] Aalami OO, Allen DB, Organ Jr CH. Chylous ascites: a collective review. Surgery 2000;128:761-78. [2] Livesey E, Davenport M. Spontaneous perforation of the biliary tract and portal vein thrombosis in infancy. Pediatr Surg Int 2008;24:357-9. [3] Leibovitch I, Mor Y, Golomb J, et al. The diagnosis and management of postoperative chylous ascites. J Urol 2002;167:449-57. [4] Andreou A, Papouli M, Papavasiliou V, et al. Postoperative chylous ascites in a neonate treated successfully with octreotide: bile sludge and cholestasis. Am J Perinatol 2005;22:401-4. [5] Lloyd DA. Gastroschisis, malrotation, and chylous ascites. J Pediatr Surg 1991;26:106-7. [6] Drinnen D, Filston HC. An unusual case of gastroschisis with gallbladder evisceration and late postrepair chylous ascites. J Pediatr Surg 1997;32:1804-5. [7] Parys SC, Hart MH. Chylous ascites following a Nissen fundoplication. J Pediatr Gastroenterol Nutr 1992;15:181-3. [8] Bhatia C, Pratap U, Slavik Z. Octreotide therapy: a new horizon in treatment of iatrogenic chyloperitoneum. Arch Dis Child 2001;85: 234-5. [9] Prabhakaran K, Vidyadhar M, Jahoorahmad P, et al. Chylous ascites following liver resection—case report. Pediatr Surg Int 2004;20: 719-21. [10] Baran M, Cakir M, Yüksekkaya HA, et al. Chylous ascites after living related liver transplantation treated with somatostatin analog and parenteral nutrition. Transplant Proc 2008;40:320-1. [11] Todani T, Watanabe Y, Urushihara N, et al. Biliary complications after excisional procedure for choledochal cyst. J Pediatr Surg 1995;30: 478-81. [12] Yamataka A, Ohshiro K, Okada Y, et al. Complications after cyst excision with hepaticoenterostomy for choledochal cysts and their surgical management in children versus adults. J Pediatr Surg 1997;32: 1097-102. [13] Yamataka A, Ohshiro K, Okada Y, et al. Early and late results of excision of choledochal cysts. J Pediatr Surg 1997;32:1563-6.