J Korean Med Sci.  2006 Feb;21(1):155-159. 10.3346/jkms.2006.21.1.155.

Trousseau's Syndrome in Association with Cholangiocarcinoma: Positive Tests for Coagulation Factors and Anticardiolipin Antibody

• Jang JW
• Yeo CD
• Kim JD
• Bae SH
• Choi JY
• Jung ES
• Rha SE
• Byun JY
• Yoon SK

• KMID: 2157800
• DOI: http://doi.org/10.3346/jkms.2006.21.1.155

Abstract

Thromboembolic events are reported to occur with a high frequency in the setting of malignancy. However, reports on an association between cholangiocarcinoma and pulmonary thromboembolism, thus far, are almost lacking. We present here an unusual case of a 56-yr-old patient presenting cholangiocarcinoma and unexplained pulmonary thromboembolism. The patient had been quite healthy before the diagnosis. Coagulation tests showed elevated levels of fibrinogen, fibrinogen degradation product (FDP), D-dimer, and IgM anticardiolipin antibody (aCL Ab). The thromboemboli were resolved 3 weeks after anticoagulant therapy using lowmolecular-weight-heparin. Then, follow-up coagulation tests showed a marked decrease to normal in aCL Ab titer as well as the normalization of FDP and D-dimer levels. In this case, we describe pulmonary thromboembolism caused by hypercoagulable state associated with cholangiocarcinoma and speculate that such a thrombotic phenomenon could be regressed by anticoagulant therapy.

Keyword

Cholangiocarcinoma; Pulmonary Embolism; Heparin; Heparin, Low-Molecular-Weight

MeSH Terms

Antibodies, Anticardiolipin/blood
Anticoagulants/therapeutic use
Bile Duct Neoplasms/*complications
*Bile Ducts, Intrahepatic
Blood Coagulation Factors/analysis
Cholangiocarcinoma/*complications
Fibrin Fibrinogen Degradation Products/analysis
Fibrinogen/analysis
Heparin, Low-Molecular-Weight/therapeutic use
Humans
Male
Middle Aged
Pulmonary Embolism/*blood/drug therapy/etiology
Syndrome
Treatment Outcome

Figure

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  Fig. 1 Abdominal dynamic CT scan show about 6.5×7×7 cm-sized and ill-defined mass with several daughter nodules in the left lobe. The huge mass with a dilatation of intrahepatic bile ducts is not enhanced on the arterial phase (A), but shows delayed enhancement on the portal phase (B), indicating cholagiocarcinoma.

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  Fig. 2 Photomicrograph of liver biopsy specimens. Moderately differentiated adenocarcinoma is shown in the hematoxylin-eosin stain (A; original magnification ×100). On the immunohistochemical staining by using cytokeratin 19 (CK 19), dark-brown staining patterns are observed on the epithelium of proliferating bile ducts (B; original magnification ×400).

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  Fig. 3 Initial chest CT scan shows (A) a filling defect with lower density in the left interlobar pulmonary artery, indicating pulmonary thromboembolism. (B) Follow-up chest CT scan after anticoagulant therapy over 3 weeks revealed the interval regression of the thromboembolism.

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  Fig. 4 Ventilation-perfusion scan reveals that no definite ventilatory abnormality in both lungs is shown (A), but multiple perfusion defects are found on the left lower lobe (B).

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  Fig. 5 Changes in FDP, D-dimer, fibrinogen, and aCL Ab levels during anticoagulant therapy. The levels of FDP, D-dimer, and aCL Ab were normalized, but fibrinogen level was slightly decreased.

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