CASE REPORT

Hepato-biliary Cystadenoma with Intraductal Extension:Unusual Cause of Obstructive Jaundice

Soumil Vyas & Sheraz Markar & Tarek Ezzat &Manuel Rodriguez-Justo & George Webster &

Charles Imber & Massimo Malago

Published online: 28 May 2011# Springer Science+Business Media, LLC 2011

Introduction

Hepatobiliary cystadenoma with mesenchymal stroma wasfirst described as a distinct histopathologic entity byWheeler et al. [1]. These potentially malignant tumours,representing a small proportion of all cystic lesions of theliver and biliary tract, occur more commonly in women ofchild-bearing age [2, 3]. Complete surgical excision is therecommended treatment in view of their malignant potential[2]. Histologically, these tumours are defined by a columnaror cuboidal epithelium, an intermediary subepithelial layerof mesenchymal or fusiform cells (ovarian-like stroma) andan external layer formed by a fibroconjunctive sheath thatseparates it from adjacent hepatic parenchyma, making a

pseudocapsule [3]. Two distinct types are recognizeddepending on the presence or absence of mesenchymal“ovarian-type” stroma [3].

We present an interesting case of a female patientadmitted with painless obstructive jaundice. Imaging andendoscopic retrograde cholangio-pancreatography (ERCP)revealed the presence of an obstructing endobiliary lesionwith extension into the liver. Histology confirmed thelesion to be a mucinous cystadenoma of the liver withovarian type stroma.

Clinical History and Findings

A 41-year-old female presented to her local hospital with ahistory of epigastric pain and obstructive liver enzymes. Aninitial ultrasound confirmed the presence of dilated intra-and extrahepatic ducts in the absence of any gall stones orcholedocholithiasis. ERCP (Fig. 1) revealed a multilobu-lated filling defect into the common hepatic duct extendinginto the left hepatic ductal system and involving the originof the right hepatic duct. An occlusion cholangiogramshowed the presence of a filling defect within the lefthepatic duct. A 3-mm balloon could be pulled past thefilling defect with relative ease. The filling defect appearedto be a soft tissue lesion occluding the left ductal systemwhich was entered with difficulty. A 9-cm 7 Fr endobiliarystent was positioned reaching proximal to the lesion withinthe bile duct. Biopsies and brush cytology from the lesionwere inconclusive and showed some inflammatory tissueand cells only, with no evidence of dysplasia or malignancy.Contrast-enhanced computerised tomography (CTscan) of thelesion showed a 5-cm well-defined cystic lesion, with internalseptations in the left lobe involving segments 4B and 3(Fig. 2). There was also bilateral dilatation of the intrahepatic

S. Vyas (*) : S. Markar : C. Imber :M. MalagoDepartment of Hepato-Pancreatico-Biliary Surgery,University College London Hospitals NHS Foundation Trust,235 Euston Road,London NW1 2BU, UKe-mail: soumil_v@yahoo.com

T. EzzatDepartment of HPB Surgery, University College Hospital,London, UK

T. EzzatDivision of Surgery, University of Alexandria,Alexandria, Egypt

M. Rodriguez-JustoDepartment of Histopathology, University College Hospital,London NW1 2BU, UK

G. WebsterDepartment of Medical Gastroenterology,University College Hospital,London NW1 2BU, UK

J Gastrointest Canc (2012) 43 (Suppl 1):S32–S37DOI 10.1007/s12029-011-9289-6

biliary radicals predominantly on the left side. Cross-sectional imaging suggested this to be a hepatic cystadenomawith involvement of the left hepatic duct (Figs. 3 and 4).Laparotomy via a reverse-L incision confirmed the presenceof a cystic lesion in the left hemiliver involving segments 4Band 3, with extension into the left hepatic duct. Aftercompleting a cholecystectomy, the common bile duct (CBD)was divided close to the duodenum, and choledochoscopyconfirmed the presence of a lesion extending into the lefthepatic duct from the CBD.

The patient underwent a left hepatectomy includingcaudate lobe excision with resection of the extrahepaticbiliary tree as a definitive surgical procedure from whichshe made a smooth and an uneventful recovery. A Roux en

Y hepaticojejunostomy was done to reconstruct the biliarytree. The final histology showed the presence of a 30×16×11-mm cystic lesion arising from the left hepatic lobe,invading the left hepatic duct. Histology confirmed thesame to be a completely resected mucinous cystadenomawith ovarian type stroma with no evidence of any dyplasiaor malignancy (Fig. 5). The patient is clinically well18 months following resection and imaging confirmed noevidence of any recurrent disease.

Methods: Literature Search Strategy

A systematic literature search of Medline (1950–October2009), Embase (1974–October 2009), Web of Science

Fig. 3 Cystic lesion within left liver lobe abutting against the lefthepatic artery

Fig. 2 CT scan showing cystic lesion in the liver and common bileduct (CBD)

Fig. 1 ERCP with fillingdefect in the mid-CBD andnarrowing of the leftintrahepatic duct

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(1990–October 2009) and Cochrane Library (2009 Issue 2)databases was performed. The search terms “BiliaryCystadenoma”, “obstructive jaundice” and MeSH headings“Cystadenoma”(MeSH), “Ovarian”(MeSH) and “Biliary”(MeSH) were used in combination with Boolean operatorsAND or OR. The electronic search was supplemented by ahand search of published abstracts from the EuropeanAssociation for Endoscopic Surgery (2007–2009), theSurgical Research Society, the Society of Academic andResearch Surgery, the Associations of Surgeons of GreatBritain and Ireland, the Association of Upper Gastro-intestinal

Surgeons and the Society of American Gastrointestinal andEndoscopic Surgeons. Reference lists of all relevant studieswere reviewed, and the search included the Current ControlledTrials Register.

Discussion

We reviewed the published literature regarding extra-hepatic(or intra-hepatic in origin with extra-hepatic extension) biliarycystadenoma. Intrahepatic biliary cystadenoma was firstdescribed by Keen in 1892 [4]. Van Gulik et al. reported on13 patients with hepatobiliary cystadenoma or cystadenocar-cinoma over a 14-year period [5]. Cases similar to ours havepreviously been reported [2, 4]. Mucinous cystadenomas ofthe biliary tract are rare unicystic or multicystic epithelialneoplasms that may arise in the liver or the extrahepatic bileducts. These cystic lesions account for less that 5% of allcystic lesions in the liver. They vary in their incidence from5–11% [6, 7]. More common cystic lesions of the liverinclude simple cysts, cystic degeneration in malignanttumours, hematomas and bilomas, abscesses, parasitic cysts(hydatid), intrahepatic choledochal cysts (Caroli disease) andbiliary cystadenomas [6]. Although 80–85% of hepatobiliarycystadenomas arise in the liver; they can occur lessfrequently in the extrahepatic bile ducts [7–9]. Origin ofthese lesions from the extrahepatic bile ducts is even rarer,

Fig. 4 Intrahepatic cystadenoma with extension into the CBD

Fig. 5 a Cystadenoma: grossappearance. b Green arrow,mucinous epithelial cells; bluebracket, ovarian stroma. cOvarian stroma; blue arrow,single stromal cells; inset bottomright corner, IHC shows stromalcell positive with oestrogenreceptor (ER)

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and less than 100 cases so far have been reported [10]. In aninteresting review Nagorney et al. from the Mayo Clinicreported on seven cases of extrahepatic biliary cystadenomasover a 43-year period [11]. The most common presentingsymptoms include epigastric pain, apart from fever, anabdominal mass and jaundice [11, 12]. Intrahepatic tumourscause jaundice by either direct tumour extension into theducts or extrinsic compression [13]. In their review ofextrahepatic bile duct mucinous cystadenomas, Nagorneyfound 96.3% of patients were female with obstructivejaundice being the presenting symptom in 85% of patients[11]. Similarly, Van Gulik reported on 15 patients withmucinous cystic tumours of the liver and biliary tract over a15-year period. One third of the patients in their seriespresented with jaundice, and nine patients were treated witha liver resection while the remaining six patients hadennucleation [3]. Mucinous cystadenomas of hepato-biliaryorigin are true epithelial proliferative tumours which areinsidiously progressive and may take many years forenlargement. However, these lesions are potentially malig-nant with a tendency to undergo malignant degeneration intocystadenocarcinomas. The risk of malignant transformationhas been quoted at less than 20% [2, 3]. Since these tumourshave the potential for malignant transformation, completionsurgical excision is recommended [2]. The lesions varyvariously in size from 2–40 cm [14]. About 80–90% of theselesions occur in women with a mean age of 45 years [14].Hence, the role of possible hormonal influence has beenconsidered due to the lesion being predominantly occurringin younger women of child-bearing age [14, 15]. Differentradiological modalities produce characteristic appearancessuggestive of biliary cystadenoma that may help guide thisdiagnosis and initiation of treatment. Ultrasound is a usefuldiagnostic modality to demonstrate hepatic cystadenomas,particularly in definition of internal morphology [16, 17].Ultrasound appearances of biliary cystadenoma typicallyinclude well-demarcated, thick-walled, noncalcified, anechoicor hypoechoic, globular or ovoid, cystic mass with internalseptations [8, 18, 19]. Contrast-enhanced CT with arterialand portal phase provides added surgical benefit in operativeplanning to determine size, morphology and anatomicalrelationship to adjacent structures, especially the bloodvessels and hence, help plan the surgical approach [20].Conventionally, cross-sectional imaging using contrast-enhanced computerised tomography has been the standardinvestigative modality for space-occupying hepatic lesions.On CT scanning, biliary cystadenomas appear as a solitarycystic mass with a well-defined thick fibrous capsule, withmural nodules, internal septa causing multiloculation and,rarely, capsular calcification [7]. Presence of septationswithin the cystic lesion helps to accurately differentiate thesame from other cystic lesions of the liver including simplecysts and choledochal cysts. Septations are diagnostically

important because they allow distinction between extra-hepatic biliary cystadenomas and choledochal cysts or cysticdilatation of the bile duct proximal to benign polyps [21].Common features of biliary cystadenomas on CT includelow-density, well-defined, thick-walled and multilocular [22,23]. Magnetic resonance cholangio-pancreatography is use-ful to establish the diagnosis and extent of hepatobiliarycystadenomas and cystadenocarcinomas [24]. Typically, onmagnetic resonance imaging (MRI), biliary cystadenomashave a homogenous low signal on T1-weighted imaging andpronounce homogenous high signal on T2-weighted imaging[25]. ERCP achieves direct visualization of the biliarycystadenoma and allows tissue sampling to obtain histologicaldiagnosis. Apart from achieving tissue diagnosis, ERCP alsoachieves effective relief of jaundice by insertion of endobiliaryprostheses [26]. The finding of a cystic mass with multipleseptations on imaging and non-opacification of an intra-luminal mass on cholangiography is characteristic of abiliary cystadenoma [11]. However, radiological differentia-tion between benign and malignant lesions is not entirelyaccurate based on imaging alone, and there no definitivediagnostic imaging criteria differentiating between benignand malignant lesions [25]. Accurate differentiation betweenbiliary cystadenomas and cystadenocarcinomas is unreliablebased on pre-operative imaging alone [1, 27, 28]. Histologi-cally, these tumours are defined by a columnar or cuboidalepithelium, an intermediary subepithelial layer of mesenchy-mal or fusiform cells (ovarian-like stroma) and an externallayer formed by a fibroconjunctive sheath that separates itfrom adjacent hepatic parenchyma, making a pseudocapsule[5]. More recently, in a review, biliary cystadenomas havebeen divided into two groups with respect to the kind oftumour stroma (presence or absence of ovarian stroma) andalso to the presence or absence of communication betweenthe cyst and the bile ducts [29–31]. Mucinous cystadenomaswith ovarian stroma almost always occur in women and maysuggest a strong correlation with ovarian mucinous neo-plasms [3]. The aetiology and origin of extrahepatic biliarycystadenomas has remained the subject of some debate forseveral years. Some authors believe cystadenomas arecongenital and arise from either aberrant harmatomatous bileducts or ectopic rests of embryonal gallbladder [1, 15]. Also,the presence of ovarian stroma (OS) in cystadenomas hasbeen correlated to the close embryological location of gonadsto the developing liver. Some of the gonadal cells maymigrate into the liver, hence contributing to the presence ofOS in these liver lesions [3, 32–34]. However, biliarycystadenomas may also be acquired neoplastic lesions, dueto their dense cellular walls and the mucinous secretions thatoccupy them [2, 22]. Histological differentiations of biliarycystadenoma from other cystic liver lesions are based on itsmultilocularity, columnar epithelium, papillary infoldings andovarian-like stroma [35]. Cystadenoma with mesenchymal

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stroma has a strong predominance in women compared withcystadenoma with hyaline stroma [36, 37]. Biliary cystade-noma represents a pre-malignant stage in the development ofcystadenocarcinoma [36, 38]. Based on previous reportedexperiences with biliary cystadenomas, extension of hepaticcystadenomas into the biliary tree is not very common. Wewould agree with previous reports that there may a specialgrowth pattern of some of these lesions, which causes them toextend into the bile ducts therefore causing jaundice [2, 6, 13,39, 40]. Ever since the first reports of successful resection ofbiliary cystadenoma were reported in 1892, several differenttreatment options ranging from simple ennucleation to localexcision and radical excision have been proposed [2, 41].Radical complete surgical resection is recommended forhepatobiliary cystadenomas and cystadenocarcinomas [11,42]. If surgical resection is complete, both biliary cystadeno-mas and cystadenocarcinomas with mesenchymal stromahave a good prognosis [43, 44]. Patients with suboptimalresection have recurrence rates of about 50% [11]. Cystade-noma with mesenchymal stroma is believed to be aprecancerous lesion, with resulting cystadenocarcinoma (withmesenchymal stroma) having a good prognosis [36]. Sincemost of the patients who have this potentially malignantlesion are middle-aged women, aggressive surgery should beoffered to all as a definitive treatment. These patients arelikely to be good anaesthetic candidates with no compellingfactors that preclude major liver surgery. Also, with the ever-increasing safety of liver surgery in centralized hepato-pancreatico-biliary units, the outcomes of liver surgery forthese patients are favourable. More importantly, we feel that,as cross-sectional imaging is done with increasing frequencydue to easy availability and more liberal use by surgeons andclinicians, akin to many other cystic conditions (e.g.pancreas), there is likely to be an increased detection ofcystic liver lesions. The presence and occurrence of biliarycystadenomas must be borne in mind and forms an importantdifferential diagnosis. This is particularly so because of theirpotentially malignant nature and the possibility of a definitivesurgical cure.

Conclusion

Hepatobiliary cystadenomas are potentially malignant lesionsand unusual causes of obstructive jaundice. Amultidisciplinaryteam approach requiring close cooperation between theradiologist, medical gastroenterologist and surgeon would berequired for successful management of this condition. CT andMRI imaging cannot, with certainty, differentiate betweenbenign and malignant forms of the cysts. Radical excisionprovides surgical cure for the condition. The condition is rare,but an aggressive management option is mandatory forsuccessful outcomes.

Acknowledgements This article and its contents have not beensubmitted simultaneously to another journal, have not been acceptedfor publication elsewhere and have not already been published.

SM and SV drafted the manuscript. All authors have significantlycontributed to, read and approved the final manuscript. All authorsdeclare that they have no competing interests. There are no conflicts ofinterest. There are no financial disclosures.

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