A case of Trousseau’s syndrome due to intrahepatic cholangiocarcinoma with an extremely high level of CA19-9

Abstract

Background: Trousseau's syndrome is a cancer-associated thrombosis. Trousseau's syndrome with cholangiocarcinoma is a rare condition with poor prognosis. Case presentation: A 59-year-old female was admitted to our hospital with abdominal pain, headache, and nausea. Abdominal enhanced computed tomography revealed liver tumor, splenic infarction, and bilateral renal infarction. Multiple acute cerebral infarctions were also detected by magnetic resonance imaging. Her preoperative serum levels of carbohydrate antigen 19-9 (CA19-9) and carcinoembryonic antigen (CEA) were > 120,000 U/mL and 589.6 ng/mL, respectively, which were extremely high. Histopathology after right hepatectomy revealed moderately differentiated adenocarcinoma consistent with intrahepatic cholangiocarcinoma. Her serum levels of CA19-9 were trending down to 9029.2 and 2659.8 U/mL at 1 and 3 weeks after surgery, respectively. However, at 7 weeks after surgery, her CA19-9 levels increased in the presence of positive imaging findings in the remnant liver, hilar lymph nodes, and peritoneal cavity. The initiation of combination chemotherapy including gemcitabine and cisplatin had a significant effect. The patient was doing well at 6 months after the surgery. Conclusion: This rare case of Trousseau's syndrome due to cholangiocarcinoma suggests that extremely high CA19-9 levels might be a pathogenic factor of this syndrome.

Nonbacterial thrombotic endocarditis as an initial presentation of advanced cholangiocarcinoma in a young patient: A case report

“…8 Since then very few cases, with a total of only 11 cases including an autopsy case, had been reported. 9–13 Among the few previously reported cases, only three cases had confirmed NBTE with echocardiography finding of valve mass or vegetations, the present case is the youngest case. 9,13,14 Cholangiocarcinoma associated with NBTE has poor prognosis and most of the reported cases were advance at diagnosis or were diagnosed on autopsy.…”

Hyperlipidemia as a risk factor for Trousseau syndrome‑related cerebral infarction in patients with advanced gastrointestinal cancer

“…Gemcitabine is a widely used anticancer drug for patients with different types of cancer, including pancreatic, biliary tract and lung cancer ( 43 ). Several case reports have demonstrated the occurrence of Trousseau syndrome under chemotherapy using gemcitabine ( 44 , 45 ). A previous study revealed that gemcitabine kills proliferating endothelial cells by activating acid sphingomyelinase ( 46 ).…”

Risk assessment and prognostic analysis of patients with splenic infarction in emergency department: a multicenter retrospective study

“…Splenic infarction in hepatocellular carcinoma was formerly reported in sorafenib use 29 , post transcatheter arterial chemoembolization 30 , and post ethanol ablation 31 . Two recent case reports revealed splenic infarction in cholangiocarcinoma, depicted as Trousseau's syndrome 32 , 33 . There appears to be a high association with the hyper-coagulation system induced by mucin-producing adenocarcinoma.…”