This is the first report of an emergent salvage surgery for lung cancer recurrence in the setting of BPAF development after PBT. Recently, the need for salvage procedures after definitive chemotherapy and/or radiotherapy for early or advanced lung cancer has been increasing, partly because there are more patients undergoing chemotherapy as a primary treatment and partly because newer effective chemotherapeutic drugs, such as immune checkpoint inhibitors, have become available. Furthermore, the number of patients undergoing high-dose radiation therapies such as stereotactic radiotherapy (SBRT), carbon ion therapy (CIT), and PBT, has also been increasing [6, 7]. For high-energy therapy, including PBT, SBRT, and CIT, an acceptable outcome was reported with a 3-year local control rate of 95.7% for stage I lung cancer [8, 9]. However, the long-term feasibility is unknown, because the inoperable patients did not survive in the long term due to poor general health conditions. One study reported that a patient with peripherally located lung cancer who had undergone SBRT developed massive hemoptysis after 4 years and was successfully treated with bronchial artery embolization [10]. Therefore, late-onset adverse events such as massive hemoptysis might be possible, such as in the present case.
Salvage surgery is generally a demanding and challenging procedure that requires experienced postoperative patient care; however, clinical experience is scarce, because the indication for salvage surgery is limited to selected patients, so as to perform a complete resection safely. There is no consensus on the indications for salvage surgery. As for the definition of salvage surgery, prior reports have described three categories [1, 11]: (I) salvage surgery for local recurrence after SBRT for early-staged lung cancer; (II) salvage procedure for local recurrence or a persistent tumor after full-dose chemoradiotherapy for locally advanced lung cancer, and (III) emergent lung resection for serious adverse events of hemoptysis, an uncontrollable lung abscess, or empyema during/after chemotherapy and/or radiotherapy for lung cancer. Based on recently published data, surgical outcomes of salvage surgeries from categories (I) and (II) have been reported; the rates of postoperative morbidity, mortality, and 5-year overall survival were 18.9–25%, 0–4.8%, and 79.5% for category (I), respectively, and 7.9–40.0%, 0–6.7%, and 40.6–53.3% for category (II), respectively [2,3,4,5,6,7,8,9]. Although the short- and long-term results of elective salvage surgeries from categories (I) and (II) were considered feasible and acceptable, the outcomes of salvage surgeries from category (III) are less clear. The results of three emergent salvage surgeries in terms of morbidity, mortality, and mean survival duration were 100%, 0%, and 13.5 ± 5 months, respectively, which were considered to be comparable to those of other types of salvage surgeries (categories [I] and [II]) [10]. In the present case, although the first aim of lung resection was palliation, the patient was able to receive chemotherapy again following the emergency lifesaving surgery and achieved survival for over a year. Therefore, we suggest that emergent salvage lung resection played an important role in controlling the serious life-threatening event of massive hemoptysis and saved the patient’s life, consequently enabling him to receive chemotherapy again.
BPAF is a rare critical event caused by various etiologies including infection, postoperative complications of pulmonary resection and transplantation, and chemotherapy [12, 13]. The clinical manifestation of BPAF is massive hemoptysis, and a delayed response may lead to fatal outcomes [12]. As for the etiology of the BPAF in the present case, both the hilar pulmonary artery and the left main bronchus were included in the irradiation field, and lung cancer recurrence was not confirmed at the left upper bronchus. Therefore, we considered that the BPAF may have arisen as an adverse effect of bevacizumab therapy and may have also been a late-onset adverse effect of high-energy therapy (PBT), which may have affected the event indirectly. The patient fortunately survived due to timely surgical intervention. The diagnosis of BPAF prior to surgery is very challenging, and in clinical practice, many patients with massive hemoptysis of unknown origin can die due to undiagnosed BPAF. Emergent salvage surgery should always be considered for massive hemoptysis due to a possible BPAF after high-energy therapy and/or chemotherapy.