Abstract
Introduction: This case report addresses the complexity of management of air leak and persisting infection in polymorbid patients. Case Presentation: A 56-year-old former marble mason presented with major hemoptysis. Chest CT revealed severe silicosis and pneumonia with an abscess in the right lower lobe and a pulmonary artery pseudoaneurysm. An open lower bilobectomy with empyema debridement was performed, and the posterior upper lobe segment was covered with a serratus anterior muscle flap. The second examination revealed persistent air leakage from the infected posterior upper lobe segment and necrosis of the muscle flap. Atypical resection of this segment was performed, and the surface of the lower part of the remnant lung was covered with a fat flap and then the omentum. The patient was discharged but was readmitted 2 weeks later due to empyema. During reoperation, a persistent infection in the remnant posterior upper lobe segment was observed in addition to a bronchopleural fistula. The only possible surgery that would cure the patient was right completion pneumonectomy. To avoid this high-risk operation, an endobronchial valve was placed intraoperatively in the posterior segment bronchus, leading to closure of the fistula and resolution of the infection. The patient recovered well and was discharged 10 days later. At the 1-year follow-up, the patient was free of symptoms and reported a good quality of life. Conclusion: This case is an excellent example of successful cooperation between an interventional pulmonologist and a thoracic surgeon to avoid right pneumonectomy in a polymorbid patient.
Right pneumonectomy should be avoided whenever possible, particularly in patients with non-tumor diseases.
(Extended) right pneumonectomy is associated with higher morbidity and mortality rates, particularly in patients with severe chronic lung disease, a poor nutritional status, and diabetes mellitus.
The placement of endobronchial one-way valves can resolve prolonged air leaks.
A lung abscess can lead to a pulmonary artery aneurysm/pseudoaneurysm.
Early and aggressive surgery is required to avoid lethal hemoptysis in patients with lung abscesses complicated by pulmonary artery aneurysms/pseudoaneurysms.
Prolonged air leak associated with pulmonary infection can persist despite repeated aggressive surgery consisting of extended lung resection and coverage using muscle and omentum flaps.
Intraoperative placement of a one-way endobronchial valve as a salvage treatment can control air leak, leading to infection resolution.
Excellent collaboration between pulmonologists and thoracic surgeons in experienced centers avoids right pneumonectomy in difficult situations.
Introduction
This case report addresses the complexity of surgical resection in treating infections in polymorbid patients with severe silicosis and calcified lymph nodes. This study describes the rare association of a lung abscess with a pulmonary artery pseudoaneurysm, for which surgical treatment is mandatory to avoid lethal hemoptysis. This case report also highlights the usefulness of an endobronchial one-way valve to control persistent air leakage after a surgical procedure. Finally, the findings indicate the high efficacy of interdisciplinarity between pulmonologists and thoracic surgeons in very difficult situations. The patient provided written informed consent to publish the case report, including the publication of images.
Case Report
We present the case of a 56-year-old patient who was a smoker, former marble worker, and stonemason. Medical history revealed a poor nutritional status, type 1 diabetes mellitus, and coronary heart disease. Because of recurrent cough for 1 year, a diagnostic setup with bronchoscopy and PET/CT was performed. A diagnosis of severe silicosis with enlarged calcified lymph nodes bilaterally was confirmed, and there were no signs of any tumors (shown in Fig. 1a, b). The patient was urgently admitted to the intensive care unit due to persistent major hemoptysis.
A chest CT scan revealed central pneumonia with an abscess in the right lower lobe and extension into the posterior upper lobe segment and empyema (shown in Fig. 2a). Interestingly, a pseudoaneurysm of the pulmonary artery to the lower lobe was described (shown in Fig. 2b). The pulmonologist called the surgeon, and a joint decision was made to perform the operation due to persisting hemoptysis. An extended open lower bilobectomy with empyema debridement and parietal pleurectomy was performed. The operation was technically challenging, and measures were implemented to avoid right pneumonectomy in this young patient. Necrosectomy with marsupialization of the part of the abscess in the posterior upper lobe segment and coverage with the serratus anterior muscle was performed. Histological examination revealed rupture of the abscess into a branch of the pulmonary artery close to several calcified lymph nodes (shown in Fig. 2c). Microbiological examination revealed gram-negative organisms suitable for chronic aspiration (Eikenella corrodens, Streptococcus anginosus, Streptococcus mitis, Prevotella loescheii, and Veillonella species). There was no suspicion of tuberculosis, and panfungal PCR and Grocott’s staining were negative. Hemoptysis stopped. On day 5, massive air leakage (1 L/min) was observed. During reoperation, persistent air leakage from the infected posterior upper lobe segment was observed in addition to necrosis of the muscle flap. Atypical resection of the posterior segment was performed, the surface of the lower part of the remnant lung was covered with a mediastinal fat flap, and a dressing soaked with povidone-iodine was applied. In addition, Prevotella species were locally identified. Second look operation was performed 3 days later. The omentum majus was used to provide complete coverage of the lung surface, since this tissue is well-vascularized and well known to stimulate healing. The air leakage stopped, and the patient was discharged 10 days later with instructions to complete antibiotic treatment consisting of amoxicillin/clavulanic acid and doxycycline for 2 weeks.
Ten days later, the patient was readmitted with empyema necessitans and wound infection (shown in Fig. 3a, b). During reoperation, necrosis of part of the omentum, persistent infection in the remnant posterior upper lobe segment, and a bronchopleural fistula were observed. Debridement and partial resection of 2 infected ribs were performed, and a povidone-iodine dressing was applied. The surgeon called the pulmonologist. Although right completion pneumonectomy was functionally possible, an endobronchial one-way valve was placed in the posterior segment bronchus to prevent prolonged air leakage (shown in Fig. 4). The valve was placed intraoperatively, and the surgeon immediately confirmed the cessation of the air leakage. The patient underwent 2 additional operations in addition to vacuum-assisted closure, the infection resolved, and the latissimus dorsi muscle was used to repair the chest wall defect. The patient recovered well and was discharged 10 days after normalization of the inflammatory parameters. The one-way valve was not removed. At the 1-year follow-up, the patient was free of symptoms and had a good quality of life (shown in Fig. 5a, b).
Discussion
This case report highlights the importance of an excellent interaction between a pulmonologist and a thoracic surgeon; first, in the management of major hemoptysis caused by perforation of an abscess in the main branch of the pulmonary artery; second, in the resolution of persistent infection and prolonged air leakage despite repeated surgical procedures. Such pseudoaneurysms have been described in case reports on infectious diseases, such as bronchiectasis, tuberculosis, or lung abscesses [1, 2]. Early and aggressive treatment is needed to avoid rupture of the pseudoaneurysm [1]. Embolization with coils has been successfully performed [2]. This would have been easier in our case because surgery on silicotic lungs is very challenging and is associated with high morbidity and mortality rates. In our case, the interventional radiologists did not consider embolization to control bleeding. Since the patient presented with major hemoptysis that had lasted for a few days, surgery was performed to control the bleeding and to treat the associated empyema.
Pneumonectomy for infectious diseases is associated with a morbidity rate of approximately 30%, mainly due to empyema, bronchial stump fistulas, severe postoperative bleeding, and sepsis [3]. We were aware of this high risk for complications. This is the reason why pneumonectomy was avoided from the beginning, and complete resection of part of the abscess in the posterior upper lobe segment was not performed. The portion of lung remaining after lower bilobectomy would have been too small to fill the chest cavity. Interestingly, in our case, all the material (muscle and omentum flaps) used to cover the lung was necrotic. These flaps were well-vascularized tissue known to promote and accelerate healing as well as control air leakage. The poor nutritional status of the patient with diabetes is very likely important. Finally, the only possible surgery to cure the patient was right completion pneumonectomy. This operation is associated with very high perioperative risks since inflammatory changes require an intrapericardial approach and, eventually, resection with extracorporeal circulation. Right pneumonectomy is well known to be associated with an increased risk of morbidity, particularly remnant pneumonectomy in cases of infection (up to 62%) [3, 4].
As a less invasive alternative to surgery, endobronchial valves have been used to control prolonged air leakage, particularly in high-risk patients [5, 6]. Smesseim et al. [7] reported a success rate of approximately 60% and a complication rate of 6%. In our case, the endobronchial valve not only led to the closure of the fistula but also to the indirect resolution of the infection. Indeed, as soon as the air leakage stopped, it was possible to repeat VAC and debridement of the supradiaphragmatic rest cavity, leading to definitive healing. In the operating room, the valve was placed in the bronchus to the posterior upper lobe segment, immediately confirming the success of the procedure. This case is an excellent example of successful cooperation between an interventional pulmonologist and a thoracic surgeon. This case underscores the need for effective interdisciplinary collaboration in such difficult situations in experienced centers.
Statement of Ethics
This retrospective review of patient data did not require ethical approval in accordance with local and national guidelines. The patient provided written informed consent for publication of the details of his medical case and any accompanying images.
Conflict of Interest Statement
The authors have no conflicts of interest to declare.
Funding Sources
No funding was received for this work.
Author Contributions
Didier Lardinois, MD, developed the study, prepared, and wrote the manuscript. Didier Lardinois, MD, and Michael Tamm, MD, conjointly discussed the best way to treat the patient. Kathleen Jahn, MD, placed the valve. Kathleen Jahn, MD; Aljaz Hojski, MD; Spasenija Savic Prince, MD; Nikolay Tsvetkov, MD; Zeljko Djakovic, MD; and Michael Tamm, MD, critically reviewed and approved the manuscript. Helga Bachmann, MSc CR, was involved in the finalization of the manuscript.
Data Availability Statement
All the data generated or analyzed during this study are included in this article. Further inquiries can be directed to the corresponding author.