Lung parenchymal metastases are common manifestations in patients with osteosarcoma; however, spread to the major airway itself is extremely rare. We present a young man who had been previously treated with surgical resection following preoperative chemotherapy and immediate postsurgical adjuvant chemotherapy for proximal tibial osteosarcoma. He developed metastasis to the major airways. The patient was treated with intraluminal radiotherapy (ILT) for the endobronchial metastasis. This is the first report of an endobronchial osteosarcoma that was treated with ILT with a complete endoscopic response. ILT provided excellent palliation in this particular case.