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DOI:10.2214/AJR.04.1560
AJR 2005; 185:1571-1572
© American Roentgen Ray Society


Case Report

Biliary Cystadenoma of the Gallbladder

Timothy B. Rooney1, Joel M. Schofer2, Mark D. Stanley1 and Steven L. Banks2

1 Department of Radiology, Naval Medical Center, Bob Wilson Drive, San Diego, CA 92134.
2 Department of Emergency Medicine, Naval Medical Center, San Diego, CA 92134.

Received October 5, 2004; accepted after revision December 6, 2004.

 
Address correspondence to T. B. Rooney (tbrooney{at}NMCSD.med.navy.mil).


Introduction
Top
Introduction
Case Report
Discussion
References
 
Biliary cystadenoma of the gallbladder is a rare disease with few reports in the literature [1, 2]. We present a case that includes the clinical course, radiologic findings on sonography and CT scan, and pathologic correlation of this unusual entity.


Case Report
Top
Introduction
Case Report
Discussion
References
 
A 38-year-old woman presented to the emergency department with a 1-day history of sharp, episodic abdominal pain, nausea, and vomiting. She had experienced prior episodes of similar pain the last 2 years previously, and all were self-limited. Examination showed a nondistended abdomen with normal bowel sounds and tenderness to palpation in both upper quadrants with no organomegaly, rebound, or guarding. No masses were noted and Murphy's sign was absent.

A complete blood count revealed a mildly elevated WBC and mild anemia (hemoglobin, 8.4 g/dL) with normal platelets and differential. She had normal liver function tests, a mildly elevated amylase of 139 U/L (normal range, 36-128 U/L), and normal lipase.

Initial imaging was an acute abdominal series, which was normal. Right upper quadrant sonography was performed that showed a thick, hyperechoic septation in the midportion of the gallbladder with apparent separation of the gallbladder into two compartments. The distal compartment contained diffuse low-level echoes while the proximal compartment was anechoic and contained multiple gallstones. The central septation itself was complex with multiple thin echogenic septa separating small hypoechoic cystic regions for approximately half of its length and a thick septation for the remainder (Figs. 1A and 1B). The gallbladder wall was otherwise not thickened, bile ducts were normal, and a small amount of pericholecystic fluid was present. The patient had no Murphy's sign on sonography.



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Fig. 1A 38-year-old woman with biliary cystadenoma of gallbladder. Transverse sonogram through gallbladder. Biliary cystadenoma is seen as rounded region of peripheral cystic spaces surrounding hyperechoic focus (arrow).

 


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Fig. 1B 38-year-old woman with biliary cystadenoma of gallbladder. Longitudinal sonogram of gallbladder. Thick septation is present, dividing gallbladder into two compartments. Biliary cystadenoma is within septation near arrow.

 
The patient was admitted and had an abdominal CT scan that showed the complex septation within the gallbladder and cholelithiasis as seen on comparison sonography (Fig. 1C). The remainder of the abdominal CT was unremarkable. Specifically, no evidence of additional intrahepatic or biliary ductal lesions was seen. The presumptive diagnosis was focal adenomyomatosis and cholecystitis. The patient later had a laparoscopic cholecystectomy without complications. Surgical pathology showed a firm, circumferential submucosal mass measuring 1.2 x 0.8 x 0.8 cm, closing off the gallbladder lumen and segregating the distal and the proximal compartments. The total gallbladder measurements were 5.0 x 3.5 x 0.5 cm. The local and Armed Forces Institute of Pathology diagnosis described smooth-muscle hypertrophy, marked hypertrophy of nerves, and multiple cysts lined by benign cuboidal epithelium with cilia consistent with biliary cystadenoma of the gallbladder.



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Fig. 1C 38-year-old woman with biliary cystadenoma of gallbladder. Axial contrast-enhanced CT image through gallbladder. Biliary cystadenoma is seen as faint septa enclosing cystic spaces (arrow).

 

Discussion
Top
Introduction
Case Report
Discussion
References
 
Biliary cystadenoma of the gallbladder is rarely reported in the literature. Simmons et al. [1] described a case in 1989 in which a 65-year-old white woman was admitted with acute-on-chronic abdominal pain, increasing girth focused in the right upper quadrant, jaundice, and fever. A clinically palpable right upper quadrant mass was found on CT and sonography to be a large cystic lesion with multiple septa emanating from the gallbladder fossa and causing biliary ductal compression. Her cystic mass was removed by open laparotomy. The imaging findings were similar to those described for intrahepatic biliary cystadenoma but originating from the gallbladder [1].

In the 2003 pathology literature, Terada et al. [2] reported a biliary cystadenocarcinoma within the gallbladder with cystadenoma elements. An 88-year-old man was admitted for right-sided abdominal pain and jaundice. Imaging at the time of admission, sonography, and CT showed a cystic tumor of the gallbladder fundus. Pathologic diagnosis was consistent with biliary cystadenocarcinoma with elements of cystadenoma, suggesting malignant transformation of cystadenoma [2].

These two previously reported cases and our case have in common right upper quadrant abdominal pain as a presenting complaint. Sonography and CT show the cystic nature of these tumors. The cystic regions in our case were smaller than in the previously reported lesions. The differential diagnosis based on the imaging findings in our case included adenomyomatosis of the gallbladder, which can present with a thick septation and cystic structures within the gallbladder wall, representing Rokitansky-Aschoff sinuses.

Although biliary cystadenoma and cystadenocarcinoma of the gallbladder have been rarely reported in the literature, both intra- and extrahepatic biliary cystadenoma and cystadenocarcinoma have been discussed more comprehensively [3-6]. The clinical symptoms are similar to those that our patient described and include intermittent abdominal pain and epigastric discomfort in middle-aged women. Sonography shows cystic masses with multiple septations and papillary projections [5, 6]. Low-density lesions (less than 30 H) with internal septa and nodules are seen on CT [6]. Although CT findings are more specific in defining the size and extent of these lesions, sonography is better able to describe the internal structure [6].

We present a rare biliary cystadenoma of the gallbladder that mimicked adenomyomatosis. Sonography and CT were complementary in determining the extent and character of the disease and can help guide the surgeon in managing this unusual entity.


References
Top
Introduction
Case Report
Discussion
References
 

  1. Simmons TC, Miller C, Pesigan AM, et al. Cystadenoma of the gallbladder. Am J Gastroenterol 1989;84 : 1427-1430[Medline]
  2. Terada T, Takeuchi T, Taniguchi M. Hepatobiliary cystadenocarcinoma with cystadenoma elements of the gall bladder in an old man. Pathol Int 2003; 53:790 -795[CrossRef][Medline]
  3. Ishak KG, Willis GW, Cummins SD, et al. Biliary cystadenoma and cystadenocarcinoma: report of 14 cases and review of the literature. Cancer 1977; 39:322 -338[CrossRef][Medline]
  4. Albores-Saavedra J, Vardaman CJ, Vuitch F. Non-neoplastic polypoid lesions and adenomas of the gallbladder. Pathol Annu1993; 28:145 -177
  5. Forrest ME, Cho KJ, Shields JJ, et al. Biliary cystadenomas: sonographic-angiographic-pathologic correlations. AJR1980; 135:723 -727[Abstract]
  6. Choi BI, Lim JH, Han MC, et al. Biliary cystadenoma and cystadenocarcinoma: CT and sonographic findings. Radiology 1989;171 : 57-61[Abstract/Free Full Text]

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