Abstract
Case Report: Two Cases of Rare Adenocarcinoma with Signet Ring Features Arising From Surgically Created Choledochoduodenal Fistula.
John Reuel F. Magan/Arlinking O. Go/Alexander T. Uy/Evan G. Ong
Metropolitan Medical Center
Significance: Cholangiocarcinoma is an uncommon adenocarcinoma with poor prognosis that arises from the bile duct epithelium. To date, there were no reported cases of cholangiocarcinoma from surgically created choledochoduodenal fistula (CDF) in the Philippines.
Clinical Presentation: Case 1: A 46/Male with a childhood history of surgically created biliary-enteric fistula developed epigastric pain, jaundice and profound weight loss from a stenotic CDF. He underwent a series of dilatation and stenting until he was stent free. Two years from the last stenting, he returned with cholangitis. Endoscopy showed an irregular nodular lesion, covered with exudates and abnormal vessels at the fistula opening. Case 2: A 54/Male presented with recurrent cholangitis secondary to cbd stone. Past history revealed a surgically created biliary-enteric fistula of 20 years. He underwent serial dilatation and stone extraction. During the last endoscopy, a firm, necrotic, friable lesion with elevated edges was seen at the fistula.
Management: Both cases had histopathologic diagnosis of Poorly differentiated Adenocarcinoma with Signet ring features. Both underwent surgery for definitive treatment. However, intraoperative findings for both patients showed far advanced and unresectable tumors.
Recommendation: To the best of our knowledge, this is the first description of cholangiocarcinoma with signet ring features from surgically created choledochoduodenal fistula in the English literature. We may recommend standard surveillance for malignant transformation for all patients with surgically created biliary-enteric fistula if it is complicated with bile stasis and recurrent cholangitis.
Keywords: choledochoduodenal fistula, cholangiocarcinoma, signet ring cells
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