Abstract

Biliary cystadenomas are rare, benign but potentially malignant, multilocular, cystic neoplasms of a biliary origin. They often present with non-specific symptoms. We present a rare case of an intrahepatic biliary cystadenoma causing luminal obstruction to the bile duct. A 32-year-old female with obstructive jaundice was evaluated and diagnosed as having a cystic lesion in the liver. With a preoperative differential diagnosis of a hydatid cyst, she underwent laparotomy and enucleation of the lesion. Intraoperatively, the tumor was found to be extending into the bile duct, which was occluding the lumen. This was excised and a bilioenteric anastomosis was done. Her post-operative period was uneventful and she was anicteric on follow-up.Biliary cystadenoma should be considered a differential diagnosis when radiologic imaging studies sug-gest a multilocular cystic hepatic lesion. Presence of jaundice in such patients should raise the suspicion of an intrabiliary component, the omission of which can lead to avoidable postoperative morbidity.

Key words: Cystadenoma, jaundice, liver, mucinous

Case Report

Institute of Surgical Gastroenterology & Liver Transplantation Centre for GI BleedDivision of HPB DiseasesStanley Medical College HospitalOld Jail RoadChennai, India

Received: December 08, 2012Accepted: December 23, 2012 Arch Clin Exp Surg 2014;3:251-253 DOI:10.5455/aces.20121223012149

Corresponding author:Dr. Ashwin RammohanInstitute of Surgical Gastroenterology & Liver Transplantation Centre for GI BleedDivision of HPB DiseasesStanley Medical College HospitalOld Jail RoadChennai, Indiaashwinrammohan@gmail.com

IntroductionBiliary cystadenomas are rare, solitary,

nearly always mucinous, multilocular, cystic tumors that can arise within the liv-er, extrahepatic bile ducts, or gallbladder [1]. The majority (80%) arise from the intrahepatic ducts and are usually asymp-tomatic [1,2]. Fewer than 100 reports of

intrahepatic biliary cystadenomas have been in medical literature, of which only 8 have presented with obstructive jaun-dice [3-5]. In this case report, we present a patient with intrahepatic biliary cystad-enoma (BCA) extending into the com-mon bile duct, presenting as obstructive jaundice.

Episodic Biliary Obstruction: Intrahepatic Biliary CystadenomaAshwin Rammohan, Jeswanth Sathyanesan, Ravichandran Palaniappan

1Bauru Dental SchoolUniversity of São PauloBauru–SP, Brazil

2Araraquara Dental SchoolSão Paulo State UniversityAraraquara-SP, Brazil

Received: February 05, 2012 Accepted: February 29, 2012 Arch Clin Exp Surg 2012;X: X-XDOI: 10.5455/aces.20120229052919

Corresponding authorÉrica Dorigatti de AvilaDepartamento de Estomatologia da Faculdade de Odontologia de BauruUniversidade de São Paulo (USP)Avenida Alameda Octávio Pinheiro Brizola, 9-75, 17012-901 Bauru–SP, Brasilerica.fobusp@yahoo.com.br

Original Article

Increased of Langerhans Cells in Smokeless Tobacco-Associated Oral Mucosal Lesions

Érica Dorigatti de Ávila1, Rafael Scaf de Molon2, Melaine de Almeida Lawall1, Renata Bianco Consolaro1, Alberto Consolaro1

Abstract

Objective: To evaluate the changes in the number of Langerhans Cells (LC) observed in the epithelium of smokeless tobacco (SLT-induced) lesions. Methods: Microscopic sections from biopsies carried out in the buccal mucosa of twenty patients, who were chronic users of smokeless tobacco (SLT), were utilized. For the control group, twenty non-SLT users of SLT with normal mucosa were selected. The sections were studied with routine coloring and were immunostained for S-100, CD1a, Ki-67 and p63. These data were statistically analyzed by the Student’s t-test to investigate the differences in the expression of immune markers in normal mucosa and in SLT-induced leukoplakia lesions. Results: There was a significant difference in the immunolabeling of all markers between normal mucosa and SLT-induced lesions (p<0.001). The leukoplakia lesions in chronic SLT users demonstrated a significant increase in the number of Langerhans cells and in the absence of epithelial dysplasia. Conclusion: The increase in the number of these cells represents the initial stage of leukoplakia. Key words: Smokeless tobacco, leukoplakic lesions, cancer, langerhans cells, chewing tobacco.

Introduction

Among tobacco users, there is a false be-lief that SLT is safe because it is not burned, which leads many people to quit cigarettes and start using SLT [1]. However, SLT con-tains higher concentrations of nicotine than cigarettes and, in addition, nearly 30 carci-nogenic substances, such as tobacco-specific N-nitrosamines (TSNA), which is formed during the aging process of the tobacco, [2-4] and which presents high carcinogenic poten-tial. Moreover, because the tobacco has direct

contact with the oral mucosa and creates a more alkaline environment, its products may even be more aggressive to tissue [5]. The percentage of SLT users is lower compared to cigarette users; however, usage is increasing among young individuals and it is therefore a significant and disturbing danger [6,7].

Initial studies on the effects of SLT on the oral mucosa demonstrated the formation of white lesions induced by chronic exposure to tobacco, characterized by epithelial thicken-ing, increased vascularization, collagen altera-

Archives of ClinicalExperimental Surgery

252 Rammohan A et al.

Case ReportA 32-year-old female presented with abdominal

pain and recurrent episodes of jaundice for the past one year. Physical examination was unremarkable, apart from icterus. Her blood investigations were nor-mal, apart from her liver function tests which showed mild elevation of total bilirubin of 3.6 mg/dL; direct component of 2.1 mg/dL. Her Serum CA 19-9 was 28.9 U/mL. Echinococcal serology was equivocal. MRI showed a well-defined cystic lesion (17.8 cm * 11.4 cm). Hypointensity on T1 and hyperintensity on T2 were seen in segments 5, 6, and 7 of the liver with few thin internal septae, suspicious of a hydatid cyst. There was no obvious intraluminal mass lesion in the bile duct observed on MRI (Figure 1). Therefore, she was planned for exploration and resection of the hydatid cyst. A non-anatomical liver resection of the lesion was done, and on examination of the distal cut

Figure 1. MRI of intrahepatic biliary cystadenoma.

Figure 2. Intraluminal tumor cast extricated from bile duct.

end of the bile duct, there was a palpable mass extend-ing onto the common hepatic duct. The bile duct was explored and a bile-stained membranous mass was noted within it. This was extricated from the bile duct, with a check choledochoscopy of the proximal biliary tree up to the second order division being done, which was found to be normal (Figure 2). Reconstruction was achieved by hepaticojejunostomy with a roux-en-Y loop of the jejunum. She had an uneventful postop-erative period. Histopathology revealed the lesion to be a cyst lined by a single layer of low cuboidal epi-thelium, with the underlying stroma resembling ovar-ian stroma in places, reported as a multilocular biliary cystadenoma. The patient was well and anicteric on follow-up.

DiscussionBCA occurs most commonly in middle-aged fe-

males [1,2]. Episodic jaundice has only been reported in eight patients in English literature [3]. Obstructive jaundice, although not always present, is the most fre-quent presenting symptom in patients with extrahe-patic cystadenomas [2,6]. On the contrary, in intra-hepatic cystadenomas, biliary obstruction is rarely the chief presenting complaint [2,6]. There are only eight reported cases of intrahepatic BCA causing obstructive jaundice, either due to protruding polypoidal masses extending into a major duct or from intracystic hem-orrhage or intraluminal mucin secretion by the tumor [3]. The most likely cause of episodic jaundice in this patient would have been the protuberant cyst extend-ing into CBD, whereby causing ductal dilatation, as re-ported by Taketomi et al.[7] BCA has two forms: the more common mucinous and the rare serous type. The former is further subdivided by the presence or absence of mesenchymal stroma. Mesenchymal stroma occurs exclusively in women [1,2,7-9]. Pre-operative diagno-sis can be difficult, but helps to strategize surgery. Bil-iary cystadenoma is commonly mistaken for a hydatid cyst, especially in endemic areas like ours. On USG and CECT, BCAs appear as focal lesions with internal sep-tae [1,2,3,10]. A preoperative percutaneous biopsy has no additional value, as it rarely produces a definitive di-agnosis [1,2,3,10]. Intraoperative choledochoscopy is useful to assess the ductal system, as exemplified in our case [10]. Laboratory investigations may be helpful for

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DOI:10.5455/aces.20121223012149

Episodic biliary obstruction

the differentiation between biliary cystadenoma and an infective cyst based on the presence of leukocytosis, positive amoebic and echinococcal serology [1,2,7,9]. Elevated cholestatic parameters are secondary to ob-struction or compression of the biliary tree [1,2]. El-evated levels of CA 19-9 (serum and cyst fluid) have been suggested by some authors to distinguish cystad-enoma from other cystic lesions [10]. Frozen sections are not very useful due to the variability in histology of cystadenomas and their inability to rule out cystadeno-carcinomas [1,2,10]. Careful histopathologic evalua-tion of the resected specimen, therefore, constitutes the only safe diagnostic modality [1,3,10]. BCA has a high rate of recurrence and a potential for neoplastic transformation in approximately 10% of cases [1,7,10]. Cystadenoma without mesenchymal stroma is known to be more aggressive, especially in men. In the past, treatment of BCA has included aspiration, marsupi-alization, internal drainage, and partial excision. The main concerns of these methods are local recurrence, malignant transformation and misdiagnosis of cancer. The ideal treatment should be complete excision of the tumor, which includes formal liver resection or wide local excision [1-3,10]. Follow-up is conducted best by performing abdominal US or a CT scan at 6-month intervals for the first postoperative year and then annu-ally [3,11].

ConclusionIntrahepatic BCA can present with episodic surgi-

cal jaundice. Biliary cystadenoma should be suspected when radiologic imaging studies suggest a multilocular cystic hepatic lesion, especially in women. Histopatho-logical examination establishes definitive diagnosis. Due to the reported malignancy potential, radical sur-gery such as wide local excision of the lesion or hepatic resection is recommended to minimize the risk of lo-cal recurrence. In all patients with intrahepatic biliary cystadenoma, the extrahepatic and intrahepatic biliary tracts should be thoroughly evaluated intraoperatively

to ensure there are no residual tumor thrombi which could lead to undue postoperative morbidity.

Conflict of interest statementThe above doctors have no conflicts of interest or

financial ties to disclose. References

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