TOPIC: Lung Cancer TYPE: Medical Student/Resident Case Reports INTRODUCTION: We describe a case of previously treated bone leiomyosarcoma (LMS) with unique presentation of distal recurrence after a prolonged latency period. CASE PRESENTATION: A 41-year-old woman with LMS of the left tibia treated with neoadjuvant chemotherapy and surgical resection nine years ago presented with shortness of breath and cough. Physical examination revealed dullness to percussion and lack of air entry over the right lung Laboratory work-up was significant for a hemoglobin of 7.6 g/dl (normal range 12.0 – 16.0 g/dl). A chest X-ray showed complete opacification of the right lung. Chest computed tomography angiogram revealed a right-sided mass with a prominent central vessel and a large pleural effusion. A thoracentesis drained 500 milliliters of serosanguinous fluid. Pleural fluid analysis showed a lactate dehydrogenase of 1,364 and protein of 4.0. Cytology was negative for malignancy. Coil embolization of the right eleventh intercostal artery was performed due to concern for active bleeding. A repeat chest X-ray showed poor lung expansion with a large amount of residual clot and fluid. Video-assisted thoracoscopic surgery was performed with biopsy of the mass and removal of two liters of clotted blood. Pathology was consistent with LMS compatible with metastasis from the patient's previously treated sarcoma. A PET scan showed an FDG-avid mass with involvement of the right chest wall, vertebra, and right posterior ribs. Surgical resection of the mass was performed. Final pathology showed high-grade LMS measuring 8.5 centimeters. DISCUSSION: Primary bone LMS is a rare malignancy of smooth muscle differentiation accounting for less than one percent of sarcoma cases. It is considered an aggressive malignancy with high rates of recurrence. Need for adjuvant therapies is generally dependent on size, stage, location, and success of tumor resection.2 Most recurrence occurs within five years of the original diagnosis with grade and size of the primary lesion conferring the greatest risk.4,5 Our patient's time to recurrence was unusual as she developed metastatic malignancy nine years from her original diagnosis of non-metastatic, low-grade LMS.This case was also unique in that the presenting complaints of dyspnea and cough were the result of a large hemothorax. Approximately 75 cases of soft tissue sarcoma with hemothorax have been documented in the literature. Only one case involving LMS was discovered during our search. Without this rare complication, our patient's malignancy may have been more advanced before its discovery. CONCLUSIONS: Primary bone LMS is a rare malignancy with high rates of recurrence. Our patient's case highlights the importance of maintaining long-term follow up and high clinical suspicion of recurrence due to the potential for prolonged latency periods. REFERENCE #1: Serrano, C., & George, S. (2013). Leiomyosarcoma. Hematology/Oncology Clinics Of North America, 27(5), 957-974. REFERENCE #2: Adelani, A., Schultenover, S., Holt, G., & Cates, J. (2009). Primary Leiomyosarcoma of Extragnathic Bone: Clinicopathologic Features and Reevaluation of Prognosis. Arch Pathol Lab Med, 133(9), 1448-1456. REFERENCE #3: Gladdy, R., Qin, L., Moraco, N., Agaram, N., Brennan, M., & Singer, S. (2013). Predictors of Survival and Recurrence in Primary Leiomyosarcoma. Annals Of Surgical Oncology, 20(6), 1851-1857. DISCLOSURES: No relevant relationships by Ian Jackson, source=Web Response No relevant relationships by Bryan Krajicek, source=Web Response No relevant relationships by Ali Nayfeh, source=Web Response No relevant relationships by Bryton Perman, source=Web Response