Adenomas of gallbladder, the majority of cases associated with cholelithiasis, appear as polypoid structures. Gallbladder adenomas are uncommon, just found in 0.5% of the cholecystectomy, and villous adenomas of gallbladder have just been reported in a few papers. ¹ Villous adenomas do not occur frequently outside the large bowel, with high potential for malignant transformation. ² We report a case of gallbladder villous adenomas in a 69-year-old patient who has systemic lupus erythematosus (SLE).

A 69-year-old female patient was admitted in Zhejiang Provincial People′s Hospital in 2011 for investigation of a gallbladder mass. She had a 15-year history of cholelithiasis (a gallstone measuring 2 cm diameter) detected by screening ultrasonography in a local hospital. No computed tomography (CT) examination had been performed before. She was also a known case of SLE diagnosed 10 years previously. Symptoms of SLE were controlled with methylprednisolone (8 mg/day) and azathioprine (100 mg/week).

On admission, her general condition was good with no history of fever, nausea, vomiting, epigastric pain, or weight loss. Tumor markers including AFP, CEA, CA125, CA19-9, CA15-3, and CA72-4 were within normal ranges, and blood chemistry tests revealed alanine aminotransferase (ALT) 22 μ/l (normal range 0-50 μ/l), aspartate aminotransferase (AST)

26 μ/l (normal range 10-52 μ/l), total bilirubin (TBIL) 16.2 μmol/l (normal range 3.4−24 μmol/L). White blood cells were 5.55 × 10 ⁹/l, and blood sedimentation rate was 17.0 mm/h (normal <20 mm/h).

Abdominal ultrasonography showed two irregular iso-echoic masses within the lumen of the gallbladder, the bigger one′s cross-section size was about 7.6 cm × 4.7 cm. There was no detectable extension through the gallbladder wall or invasion of adjacent structures Figure 1. CT showed two broad-based cauliflower-like masses in the gallbladder, from which the bigger mass was about 7.4 cm × 5.1 cm, and the smaller one 4.1 cm × 3.6 cm. The lesions measured 31-36 Hounsfield Unit on non-contrast CT and showed intense enhancement after administration of iodinated contrast material Figure 2. There was no evidence of extra-gallbladder extension or metastatic disease. Figure 1

US shows intraluminal irregular masses (arrow). No extension through the gallbladder wall noted

Villous adenoma is a subtype of adenomatous polyps with malignant potential. Grossly, they appear as polypoidal masses with predominant villous elements. Adenomas of the gallbladder are uncommon and they are found in 0.5% of cholecystectomy specimens. They occur primarily in women, the majority of cases (50-65%) are associated with cholelithiasis, usually asymptomatic and discovered incidentally during a radiological evaluation for abdominal pain. ¹ Villous adenomas are very rarely seen in the gallbladder. These lesions are most frequently in the rectum and colon. ²

The histopathology and gross appearance of villous adenomas in the gallbladder and the gastrointestinal tract are similar. In 533 cases of cholecystectomy and 1300 randomly selected autopsies (mainly elderly individuals), villous adenomas were found in two resected cases (0.38%) and in one autopsy case (0.08%). ³ There are some reports of villous adenomas in the biliary tree and they are mostly located at the ampula of Vater. ⁴ , ⁵ Another two cases of gallbladder villous adenomas have been reported. One case which showed the lesion only by ultrasonography was in a patient with acromegaly, ⁶ and the other case was in a patient without other significant medical history, whose ultrasonography was negative and CT depicted the lesion.- ⁷ These two villous adenomas were smaller than that in our report. In the current case, we had ultrasonography, CT, and even MRI as well as histology.

Villous adenoma is currently considered a premalignant condition. The following observations support the hypothesis of adenoma−carcinoma sequence of gallbladder carcinogenesis: (1) the presence of histological and genetic transition from adenoma to carcinoma, (2) close correlation between the size of the lesion and malignant transformation, (3) orderly increase in age in the three groups, with a mean of 50 years for benign adenomas, 58 years for adenomas with malignant change, and 64 years for invasive carcinoma. ⁸ , ⁹ In a report of four patients with biliary villous adenomas (biopsy or surgically proven), two patients (50%) were subsequently found to have malignant lesions 3 years and 8 years following definitive diagnosis. ¹⁰ In addition, Kimura demonstrated positive immunohistochemical staining (CA19-9) in three patients. He also suggested that villous adenomas should be treated as a premalignant entity. ³

As ultrasonography and CT technology improves, polypoid lesions are easily found. Ultrasonography takes advantage of depicting the gallbladder wall architecture exactly, but is easily interrupted by gas and too much fat. CT seems less sensitive in identifying the different layers of the wall. ¹¹ MRI can show the surrounding structures better, and without ionizing radiation. The imaging characters of villous adenoma in the gallbladder are just like those in rectal and colon, duodenum, or bile ducts, showing soft tissue echo on ultrasonography, iso-hypodensity with various enhancing patterns on CT, while hypointense on the non-contrast T1-weighted images and heterogeneously hyperintense on T2-weighted images. ¹ , ⁵ , ¹¹ , ¹² SLE is a multisystem autoimmune disease associated with significant morbidity and mortality. ¹³ The opinions about the relationship between SLE and the gallbladder disease are different. Some authors assume that the SLE patients are prone to biliary sludge and stone formation because of biliary dyskinesia like other collagen vascular diseases. ¹⁴ Other authors think gallbladder disease is no more common in SLE patients than that in the general population. ¹⁵ There are no previous reports on the relationship between gallbladder villous adenoma and SLE. The coexistence of SLE and gallbladder villous adenoma is thought to be probably coincidental in our case.