Multiple early bile duct carcinoma associated with congenital choledochal cyst

Yoshikane, Hiroaki; Hashimoto, Senju; Hidano, Hitoshi; Sakakibara, Akira; Ayakawa, Tadao; Mori, Sumito; Kojima, Yoshihiro

doi:10.1007/s005350050113

Cited by 16 publications

(8 citation statements)

References 6 publications

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“…The malignant change occurs mainly within the choledochal cyst, but may occur in the gallbladder, pancreatic duct, and intrahepatic bile ducts 3,4 . Multiple malignant transformations are also reported 4,5,10 . In the present case the carcinomatous transformation was located within the choledochal cyst, and no carcinomatous transformations were recognized in the pancreas and gallbladder.…”

Section: Discussionsupporting

confidence: 42%

“…In the present case the carcinomatous transformation was located within the choledochal cyst, and no carcinomatous transformations were recognized in the pancreas and gallbladder. Most of the carcinoma associated with congenital choledochal cyst is adenocarcinoma, 3–6 although squamous cell carcinoma and sarcomatoid carcinoma have been reported 7–9 . Okamura et al .…”

Section: Discussionmentioning

confidence: 99%

“…Recent Japanese studies have suggested that congenital choledochal cyst is frequently associated with anomalous pancreatico‐biliary ductal union 3,4 . In the English‐language literature most of the carcinomas in congenital choledochal cyst are adenocarcinomas, 3–6 although squamous cell carcinoma and sarcomatoid carcinoma have been reported 7–9 . Only one case of adenosquamous carcinoma associated with choledochal cyst has been reported in the English‐language literature 2 .…”

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confidence: 99%

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Adenosquamous carcinoma in a congenital choledochal cyst associated with pancreatico‐biliary maljunction

Terada

2009

Pathology International

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Congenital choledochal cyst is occasionally complicated by carcinomatous transformation, mostly adenocarcinoma. Adenosquamous carcinoma arising in a congenital choledochal cyst is very rare. The author herein reports an adenosquamous carcinoma arising in congenital choledochal cyst associated with pancreatico-biliary maljunction. A 34-year-old man with congenital choledochal cyst and recurrent cholangitis had been followed up, and was admitted to hospital to undergo testing for cancer. Imaging modalities including computed tomography, magnetic resonance imaging and endoscopic retrograde cholangiography showed an elevated lesion in the choledochal cyst. Because clinical cytology of bile indicated malignant cells, pancreatico-duodenectomy, cholecystectomy, and resection of the choledochal cyst were performed. Grossly, the choledochal cyst was type I, and its size was 8 x 10 cm. Anomalous pancreatico-biliary ductal union was recognized. An elevated lesion was recognized in the choledochal cyst. Histologically, the lesion was composed of a squamous cell carcinoma element and an adenocarcinoma element; a gradual transition was recognized between the two. The squamous cell carcinoma element contained microcytic cells with mucins. On immunohistochemistry the adenocarcinoma element and microcytic cells were positive for CEA, but the squamous cell carcinoma element was negative for CEA. Both elements were positive for CA19-9. Ki-67 labeling was 53% in the adenocarcinoma element and 48% in the squamous cell carcinoma element. p53 protein was negative in both elements. At the time of writing, the patient was alive after 25 months without recurrence or metastasis. The present case is the second case of adenosquamous carcinoma arising in congenital choledochal cyst in the English-language literature.

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Section: Discussionsupporting

confidence: 42%

Section: Discussionmentioning

confidence: 99%

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confidence: 99%

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Adenosquamous carcinoma in a congenital choledochal cyst associated with pancreatico‐biliary maljunction

Terada

2009

Pathology International

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“…[13][14][15][16][17][18][19][20][21][22] (Note that the form of choledochal dilatation shown in Table 1 is according to Todani et al 23 ). The present patient represents the first reported case of triple carcinomas of the biliary tract, including lesions in the bile duct.…”

Section: Discussionmentioning

confidence: 99%

Triple carcinomas of the biliary tract associated with congenital choledochal dilatation and pancreaticobiliary maljunction

Okamura¹,

Hayakawa²,

Kuze³

et al. 2000

Journal of Gastroenterology

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We report a rare case of triple carcinomas of the biliary tract associated with congenital choledochal dilatation (CCD) and pancreaticobiliary maljunction (PBM). The patient was a 58-year-old Japanese man who complained of epigastralgia. Ultrasonography and computed tomography revealed an elevated lesion inside the markedly dilated extrahepatic bile duct, thickening of the gallbladder wall, and small polypoid lesions in the gallbladder. Magnetic resonance cholangiopancreatography and endoscopic retrograde cholangiopancreatography showed CCD and PBM. With a diagnosis of carcinoma of the bile duct and cholesterol polyps in the gallbladder, pylorus-preserving pancreaticoduodenectomy was performed. The resected specimen showed two elevated lesions in the dilated bile duct, cholesterol polyps, and an area of irregular mucosa in the gallbladder. Histopathological examination showed two carcinomas in the bile duct, an adenosquamous cell carcinoma, and a moderately differentiated tubular adenocarcinoma, and a well differentiated tubular adenocarcinoma of the gallbladder. Two years and 6 months after the operation, a solitary metastatic liver tumor was detected. Left hepatic lobectomy was performed. At present, 7 months after the second operation, the patient is doing well with no signs of recurrence. Multiple carcinomas in the biliary tract associated with CCD and PBM, including the details in the present patient, were reviewed.

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“…Because of the risk of malignant change in choledochal cysts, the ideal treatment is excision of the entire dilated biliary tree with reconstruction using a Roux‐en‐Y hepaticojejunostomy [3, 9, 10, 17]. We present a contemporary cohort of adult patients with choledochal cysts, which emphasises the complexity and diversity of this disorder in adults.…”

Section: Introductionmentioning

confidence: 99%