Extremely rare case of H-type gallbladder duplication coexistent with carcinoma: a case report and review of the literature

Abstract

Introduction: Multiple gallbladders represent a rare congenital disorder, and coexistence with carcinoma is extremely rare, leading to a high possibility of misdiagnosis and surgical complications. In this study, a case was reported and the literature was reviewed.

Case presentation: An 80-year-old woman was diagnosed with acute cholecystitis via ultrasonography and was successfully treated with antibiotics. After the patient's biliary colic relapsed, she was referred to our hospital. Multiple imaging modalities revealed duplication of her gallbladder (H-type) and suggested coexistence with carcinoma. According to preoperative evaluations, we assumed the patient had stage IIIA disease, and cholecystectomy, cholangiography using a near-infrared ray vision system, and sectionectomy of segments 4a and 5 were performed. Contrary to the high standardized uptake values obtained by ¹⁸F-fluoro-2-deoxy-D-glucose positron emission tomography, gallbladder carcinoma was pathologically diagnosed as stage 0 mucosal cancer. Seven days after the operation, portal thrombosis of the posterior branch was revealed, and conservative therapy was indicated; satisfactory results were achieved. The patient was discharged 65 days after surgery. No recurrence was observed for 1 year after surgery.

Conclusions: An extremely rare case of malignancy in a duplicated gallbladder was reported, and the literature was reviewed. Accurate estimations are feasible for diagnoses of multiple gallbladders, where correct evaluations are vital, especially in malignant cases. Because of the possibility of malignancy, resected accessory gallbladders should be scrutinized pathologically.

Keywords: Adenocarcinoma; FDG-PET; Gallbladder duplication; Intraoperative cholangiography; Near-infrared ray vision system.

Fig. 1

Ultrasonography imaging. The arrow shows carcinoma 1 cm in diameter in the proximal gallbladder with a base and obscure demarcation along the wall

Fig. 2

Abdominal enhanced CT imaging. Carcinoma in the proximal gallbladder, which was identified as the accessory gallbladder, shown by enhanced CT images. a Axial view. The low-density mass with a diameter of 1 cm was slightly enhanced (see the arrows). Direct liver invasion was less conspicuous on CT imaging. b Coronal view, and c sagittal view. Two separate cavities were clearly demonstrated

Fig. 3

MRCP imaging. a 3D image and b 2D image. A definite H-type duplicated gallbladder was demonstrated. Both cystic ducts were identified as diverging individually from the common bile duct (see the arrows)

Fig. 4

EUS imaging. Endoscopic ultrasonography demonstrated a solid tumor in the proximal gallbladder that seemed to invade the hepatic parenchyma adjacent to the gallbladder (see the arrows). The dotted line indicates the size of the mass

Fig. 5

PET imaging. PET showed an abnormal FDG uptake (SUVmax 5.97) in the gallbladder (see the arrow). No other metastases were identified

Fig. 6

Intraoperative findings. a A double gallbladder was clearly identified. b A pair of cystic ducts (indicated by arrows) were confirmed

Fig. 7

Intraoperative cholangiography. a Normal cholangiogram via iodine contrast agent. b, c Biliary images via a near-infrared ray vision system (Photo Dynamic Eye®: PDE). An intraoperative cholangiogram through the primary cystic duct showed the correct ductal anatomy. A pair of cystic ducts (indicated by arrows) were demonstrated

Fig. 8

Macroscopic findings and histological findings of resected specimen. a Resected fresh specimen, proximal gallbladder identified as an accessory gallbladder containing carcinoma (single arrow), and distal gallbladder identified as a primary gallbladder (double arrows). b Formalin-fixed specimen, carcinoma in the proximal gallbladder identified as an accessory gallbladder (arrow). c, d Hematoxylin and eosin (H&E) staining at original magnification (× 20 and × 100). Pedunculated and non-invasive mucosal carcinoma had a fine stalk, and there was space between the mass and the gallbladder wall