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Spontaneous pneumothorax is a rare complication of chemotherapy in the treatment of lung neoplasms. It is relatively common in osteosarcoma lung metastases patients following chemotherapy, but the cause is still unknown. Here, we report a case of bilateral spontaneous pneumothorax occurring in a 40-year-old female in whom lung metastases from osteosarcoma were treated with combination chemotherapy. The patient had osteosarcoma on the right side of the mandible, and developed bilateral spontaneous pneumothorax. Through contrasting chest computed tomography, pneumothorax was identified at the site of the pulmonary metastases and occurred following the formation of bullae. The pneumothorax and bullae were produced after the patient received chemotherapy. In October 2014, the patient succumbed to the disease due to hemorrhage of the primary osteosarcoma lesions.