2019 Volume 41 Issue 6 Pages 640-645
Background. Primary angiosarcoma of the bone is an extremely rare form of vascular bone tumor, accounting for <1% of primary malignant bone tumors. Its histological findings are characterized by formation of anastomosing blood vessels lined by endothelial cells. It tends to involve the long, tubular bones of the extremities, and more than half of cases are found with metastasis, especially to the lung. Once metastasis to the lung has occurred, patients usually present with symptoms such as dyspnea, chest pain, and hemoptysis. Case. An 80-year-old woman was referred to our hospital for the further evaluation of abnormal X-ray findings of her right humerus, with only slight range of motion restriction. Upon arrival, she was also diagnosed with anemia, thrombocytopenia, and elevated D-dimer levels, which satisfied the criteria for disseminated intravascular coagulation. In addition, chest X-ray showed diffuse infiltrative shadows in both lungs, and chest computed tomography (CT) showed bilateral consolidation with ground-glass opacity. The further investigation of the lungs was prioritized over her shoulder pain, and bronchoalveolar lavage was performed. The fluid collected was fresh and blood-like and contained hemosiderin-laden macrophages, confirming the diagnosis of diffuse alveolar hemorrhaging. In addition, a biopsy of the right humerus was performed at the site where magnetic resonance imaging showed a destructive lytic lesion with irregular borders of fractured bone. Immunohistochemical staining revealed the tumor cells to be positive for cluster of differentiation 31 and 34 and negative for cytokeratin and thyroid transcription factor-1, and she was diagnosed with angiosarcoma. Conclusion. We herein report a rare case in which angiosarcoma was coincidently diagnosed in connection with diffuse alveolar hemorrhaging without CT findings suggesting lung metastasis. Neoplastic diseases should not be forgotten as differential diagnoses of diffuse alveolar hemorrhaging.
