Journal of Indian Association of Pediatric Surgeons
Journal of Indian Association of Pediatric Surgeons
                                                   Official journal of the Indian Association of Pediatric Surgeons                           
Year : 2014  |  Volume : 19  |  Issue : 1  |  Page : 44--45

Benign papillomatosis of common bile duct in children: A rare case report


Amit Singh1, Nitin Sharma1, Shasanka Shekhar Panda1, Minu Bajpai1, Manisha Jana2,  
1 Department of Pediatric Surgery, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, India
2 Department of Radiodiagnosis, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, India

Correspondence Address:
Minu Bajpai
Professor of Paediatric Surgery, All India Institute of Medical Sciences, New Delhi - 110 029
India

Abstract

In children benign neoplasms are rare events in the extrahepatic biliary ducts and scanty literary references on the subject are available. We are reporting a rare case of a 3-year-old female child having biliary papillomatosis in lower common bile duct.



How to cite this article:
Singh A, Sharma N, Panda SS, Bajpai M, Jana M. Benign papillomatosis of common bile duct in children: A rare case report.J Indian Assoc Pediatr Surg 2014;19:44-45


How to cite this URL:
Singh A, Sharma N, Panda SS, Bajpai M, Jana M. Benign papillomatosis of common bile duct in children: A rare case report. J Indian Assoc Pediatr Surg [serial online] 2014 [cited 2019 Dec 10 ];19:44-45
Available from: http://www.jiaps.com/text.asp?2014/19/1/44/125966


Full Text

 Introduction



Benign neoplasms in the extrahepatic biliary ducts are rare in children. Their importance lies in their ability to mimic malignant lesions in these locations. Commonest presenting symptoms are pain, jaundice, and acute cholangitis. These cases are often diagnosed as intraoperative surprises. We are presenting our experience with one such case.

 Case Report



Our case was a 3-year-old female child who presented to our pediatric surgical outpatient department with complaints of pain in the upper part of the abdomen along with jaundice and fever for last 6 months. On per abdominal examination no lump or hepatosplenomegaly was found. With the clinical possibility of a choledochal cyst, ultrasonography (USG) along with liver function test and routine hematological investigations were advised. USG showed impacted worm-like appearance in the lower common bile duct (CBD) with proximal dilatation [Figure 1]. Choledochal cyst was ruled out. In view of the deepening jaundice, child was advised endoscopic retrograde cholangiopancreatography (ERCP) to clear the worm load in the lower end of CBD and to place the stent for establishing the bile flow. Thus, endoscopic sphincterotomy with placement of 10 Fr stent was done [Figure 2]. There were no worms in the CBD in this patient. The child also received anthelmintics. Following stent placement child recovered from jaundice within 2 weeks. As there was a doubt of persistent worm in the distal CBD, exploration was planned; and after obtaining informed written consent, the child was explored under general anesthesia. Right upper transverse incision was given, abdominal cavity was opened, liver mobilized, gall bladder identified, and CBD traced using stent as a guide. On opening the CBD multiple polyps were found with stent in situ [Figure 2] without stone or worm. In view of possible malignancy a frozen section was sent and when confirmed to be benign, polyps were removed completely along with stent and CBD was repaired over a T-tube. Postoperatively T-tube was removed after 3 weeks. The child recovered uneventfully and presently she is in close follow-up and doing well. Specimen sent for histopathological examination showed adenomatous polyps with colonic metaplasia.{Figure 1}{Figure 2}

 Discussion



Benign tumors and tumor-like lesions of the gallbladder and bile duct have a wide spectrum and despite this diversity these lesions share common embryologic origins and histological characteristics. Although in children these lesions are relatively uncommon, their importance lies in their ability to mimic malignant lesions in these locations. Benign neoplasms are derived from the epithelial and nonepithelial structures that compose the normal gallbladder and bile ducts. The epithelium gives rise to adenomas, cystadenomas, and the unusual condition of biliary papillomatosis. [1] Granular cell tumors, neurofibromas, ganglioneuromas, paragangliomas, and leiomyomas are examples of benign tumors that may originate from nonepithelial structures. [1] Biliary papillomatosis is rare with only 140 cases reported in literature that too in adults. [1] Chappet in 1894 reported the first case of biliary papillomatosis. [2] The malignant potential of benign polypoid lesions of the extrahepatic biliary system is controversial and not fully understood. The current literature supports the notion of malignant transformation of these benign epithelial lesions. [3],[4],[5] Reported male:female ratio is 2:1 with peak incidence in the 4 th decade of life. [3],[4] Commonest presenting symptoms are pain, jaundice, and acute cholangitis. [3],[4],[6] Diagnosis of these cases predominantly is intraoperative with few cases being diagnosed through ultrasound. [7] Most of them are isolated case reports only. [8] All case reports are from adult literature. Despite extensive PubMed and Medline search we were not able to find a single case report on this topic in children.

 Conclusion



From this case report we can conclude that although rare, biliary papillomatosis should be kept in mind when examining a patient with obstructive jaundice, acute cholangitis, and abdominal pain in children.

References

1Vassiliou I, Kairi-Vassilatou E, Marinis A, Theodosopoulos T, Arkadopoulos N, Smyrniotis V. Malignant potential of intrahepatic biliary papillomatosis: A case report and review of literature. World J Surg Oncol 2006;4:71.
2Chappet V. Cancer epithelial primitif du canal choledoque. Lyon Med 1894;76:145-57.
3Yeung YP, AhChong K, Chung CK, Chun AY. Biliary papillomatosis: Report of seven cases and review of English literature. J Hepatobiliary Pancreat Surg 2003;10:390-5.
4Lee SS, Kim MH, Lee SK, Jang SJ, Song MH, Kim KP, et al. Clinicopathologic review of 58 patients with biliary papillomatosis. Cancer 2004;100:783-93.
5Chae BW, Chung JP, Park YN, Yoon DS, Yu JS, Lee SJ, et al. Villous adenoma of the bile ducts: A case report and a review of the reported cases in Korea. Yonsei Med J 1999;40:84-9.
6Ma KF, Iu PP, Chau LF, Chong AK, Lam HS. Clinical and radiological features of biliary papillomatosis. Australas Radiol 2000;44:169-73.
7Bondestam S, Kivilaakso EO, Standertskjöld-Nordenstam CG, Holmström T, Hästbacka J. Sonographic Diagnosis of a bile duct polyp. AJR Am J Roentgenol 1980;135:610-1.
8Chang YT, Wang HP, Sun CT, Chang MC, Chang YC, Wu MS, et al. Papillary adenoma of the bile duct. Gastrointest Endosc 2001;53:777.