Letters to the Editor
To the Editor:
Lung is the most frequent site for metastasis from osteosarcoma. Usually, the presentation is with well-defined parenchymal nodules, and unusually with pleural deposits, calcification, or mediastinal nodes.1 Metastases from osteosarcoma presenting as an isolated endobronchial lesion is discretely rare. We recently encountered a case of small cell osteosarcoma with recurrent hemoptysis and an isolated endobronchial metastasis.
A 29-year-old man presented in August 2010 with complaints of intermittent hemoptysis of 6-month duration. He had a past history of an osteosarcoma involving shaft of left humerus that was treated in 2007 with chemotherapy, limb salvage surgery, and radiotherapy. Radiotherapy was administered as he was initially diagnosed as having Ewing sarcoma. At the time of presentation his performance status was ECOG 2. Chest computed tomography scan revealed multiple nodules in both the lungs, which were suggestive of metastases. Bone scan showed metastases to L3-L4 vertebrae. A pathology review of baseline biopsy slides confirmed a diagnosis of small cell osteosarcoma. He was started on chemotherapy consisting of alternating courses of Cisplatin/Doxorubicin and Ifosfamide/Etoposide. After 3 cycles, his hemoptysis had resolved and a complete response was achieved with resolution of lung nodules. However, after 6 cycles, he experienced recurrence of hemoptysis. Computed tomography scan revealed multiple thin-walled cavities in right lower lobe with surrounding ground glass opacities; however, no distinct nodule was noted (Fig. 1A). The hemoptysis was attributed to metastases even though radiologically metastases were not obvious. Right bronchial artery embolization was performed and the chemotherapy was changed to high-dose methotrexate. Despite an initial transient response to this therapy, hemoptysis persisted. A flexible bronchoscopy revealed a large infiltrative growth completely occluding the right main bronchus (Fig. 1B), biopsy from which showed features of metastatic osteosarcoma with large areas of necrosis (Figs. 1C, D). In view of endobronchial metastases, local therapy in the form of intraluminal brachytherapy was contemplated along with gemcitabine-based chemotherapy; however, the patient unfortunately succumbed to a spell of massive hemoptysis.
Of pulmonary sites of metastases from different tumors, spread to the major airways is extremely rare. The incidence of such metastasis is estimated to be approximately 2%; the most common primary malignancies being renal cell, colorectal, cervical, breast carcinoma, and malignant melanoma.2 Previous reports in the literature of endobronchial metastases from osteosarcoma are generally described with simultaneous lung parenchymal lesions3 and predominantly with obstructive features.4,5 Our report highlights recurrent hemoptysis that persisted despite resolution of parenchymal nodules as a feature of endobronchial pulmonary metastases.
To our knowledge, this is the first case of small cell osteosarcoma with isolated endobronchial metastases without obvious concurrent parenchymal involvement. Thus, inexplicable pulmonary manifestations should prompt an early evaluation including bronchoscopy and early institution of local therapy for possible palliation.
Smita Kayal, MD*
Anant Mohan, MD†
Sanjay Thulkar, MD‡
Prashant Durgapal, MD§
Sameer Bakhshi, MD*
*Department of Medical Oncology
§Pathology, Dr B. R. A. Institute Rotary Cancer Hospital, All India Institute of Medical Sciences, New Delhi, India
1. Rastogi R, Garg R, Thulkar S, et al. Unusual thoracic CT manifestations of osteosarcoma: review of 16 cases. Pediatr Radiol. 2008;38:551–558
2. Braman SS, Whitcomb ME. Endobronchial metastases. Arch Intern Med. 1975;135:543–547
3. Kiryu T, Hoshi H, Matsui E, et al. Endotracheal/endobronchial metastases: clinicopathologic study with special reference to developmental modes. Chest. 2001;119:768–775
4. Mogulkoc N, Goker E, Atasever A, et al. Endobronchial metastasis from osteosarcoma of bone: treatment with intraluminal radiotherapy. Chest. 1999;116:1811–1814
5. Rubio ER, Chang EE, David O, et al. Pulmonary osteosarcoma: endoscopic management. J Bronchol. 2001;8:277–281