Abstract

INTRODUCTION: Cholangiocarcinoma (CCA) is a malignant tumor which originates from the epithelial cells of bile ducts. Abdominal pain and jaundice are the most common presenting symptoms. It commonly metastasizes through lymphatics to regional lymph nodes, followed by involvement of liver, lungs and peritoneum via blood. Bone metastasis is very rare although it has been reported. We are presenting an unusual case with initial presentation of appendicular bony metastatic disease without any GI symptoms. CASE DESCRIPTION/METHODS: 59-year-old non-smoker male with past medical history of obstructive sleep apnea initially presented to ER with right arm pain while standing from a chair and felt crack in his arm. Physical exam was unremarkable except right mid arm mild swelling and tenderness. Right arm x-ray showed an acute comminuted fracture of proximal humeral shaft with minimal displacement. A CT scan was obtained due to the low energy nature of this injury to rule out pathologic etiology, but this was negative. Blood work up was positive only for normocytic anemia with hemoglobin of 10 g/dl. He was placed in a sling and scheduled to follow-up with orthopedics as outpatient. Four weeks follow up x-ray showed persistent fracture and prominent bone resorption concerning for its pathological nature. Patient was referred to oncologist. Patient remained asymptomatic except right arm pain and some swelling. Extensive work up was initiated. Laboratory investigation showed elevated LFTs with cholestatic pattern. CT CAP with contrast showed significant metastatic abdominal lymphadenopathy, ill-defined mass in the right lobe of the liver and splenomegaly. A bone scan showed uptake in the right mid humerus but no additional findings. Patient underwent CT guided bone biopsy which was consistent with metastatic intrahepatic CCA. Given aggressive growth in short duration and refractory pain, intralesional debulking and metallic end prosthetic segmental reconstruction of the right humeral diaphysis was performed followed by radiation therapy. Oncology recommended to start chemotherapy although prognosis is grim. DISCUSSION: CCA is an aggressive tumor with poor prognosis. The pattern of metastasis is usually through lymphatics and further spread via hematogenous route. Appendicular bone metastasis is very rare phenomena and only few cases reported so far. Physicians should have low threshold to investigate low energy injuries leading to bone fractures, in depth for pathological fractures and possibly underlying malignancy.

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