A 22-year-old male patient had been occasionally experiencing left chest pain and dyspnea for several years, but the symptoms had resolved spontaneously, and he had no history of medical examination. He presented to his home physician with persistent complaints of respiratory distress and high-grade fever for one month after the onset of left chest pain and dyspnea, and was found to have a pneumohydrothorax on chest radiography (Fig. 1A). For the failure of conservative treatment, he underwent thoracoscopic decortication at a local hospital (Fig. 1B). The apical part of the left upper lung, which could not be peeled off, was resected. A pathological finding of Aspergillus organisms in the pleura (Fig. 1C and D), and a serological positiveness of Aspergillus antibodies led to the diagnosis of spontaneous pneumothorax and Aspergillus empyema. The patient had no history of pulmonary aspergillosis, and no organisms were present in the lung parenchyma in the lung resection specimen. Laboratory and physical findings were negative for the presence of any disease that could cause opportunistic infections, and there was no history of any other noteworthy illness since birth and no family history of immunodeficiency diseases. It was strongly inferred the patient had developed a chronic pneumothorax cavity as a result of repeated pneumothoraces, although there was no history of the other diseases that could form cavitary lesions, such as tuberculosis. Just before the onset of symptoms, his apartment had been closed for an extended period for the summer vacation, and upon his return, he discovered that his place was heavily infested with mold due to the high summer temperatures and humidity. It was presumed that the patient developed the current pneumothorax in this moldy environment, resulting in the dissemination and infection of Aspergillus in the previously existing chronic pneumothorax cavity through the bronchopleural space. The postoperative course after the decortication and the wedge resection was good, and he was discharged home on the eighth postoperative day. However, one month after the operation, re-infection occurred and persistent air leak developed, which prompted admission to our hospital. Chest computed tomography (CT) revealed a large apical pleural space in the left without a fungus ball, and an irregularly thickened pleura in the upper thoracic cavity, suggesting the infection of the pleura and air space (Fig. 2). Pleural fluid culture detected Aspergillus fumigatus, and serum Aspergillus antibodies were again positive. Other findings on CT included a very mildly infiltrated left upper lung parenchyma with intrapulmonary infection at the bottom of the cavity. The patient had no evidence of intrapulmonary infection on imaging or histology at the time of the initial surgery, suggesting that the fungus in the pleural and pneumoperitoneum had spread to the lungs after the previous surgery; hence, the patient was diagnosed with apical pleural aspergillosis because the main body of infection was located in the pleura and airspace.
It was determined that surgical intervention for the infection site in the pleura was necessary for curative treatment. Since the intrapulmonary infection was controlled with antifungal medication, lobectomy with extensive muscle filling was considered an excessive intervention. Because the dead space was confined to the apex of the lung, we decided to perform cavernostomy and minimal dead space filling as the first-line procedure.
A 12-cm anterolateral incision was made in the third intercostal space, and cavernostomy was performed on the Aspergillus cavity, providing a clear view from apex to bottom (Fig. 3). The thickened parietal pleura, forming the cavity wall, was deemed unresectable, while the thickened lung parenchyma at the cavity bottom maintained compliance. To control infection, the pyothorax cavity was scraped with a sharp curette to remove Aspergillus fragments, and the alveolar fistula was closed by suturing the pleura as a pledget. The cavity volume was estimated at 90 mL. The extraperiosteal air-plombage method (Kinchu method) was used to minimize cavity volume. Intercostal muscle and partial serratus anterior muscle transposition flaps, attached to the second and fourth ribs, were guided into the cavity. Two thoracic drains were placed in the cavity, one outside the pleural tent, and the wound was closed.
Postoperatively, the thoracic drain was maintained for three weeks while the patient underwent intravenous voriconazole (VRCZ) therapy (4 mg/kg every 12 hours for 4 weeks), which was eventually transitioned to oral VRCZ (400 mg/day) for approximately a year. One year after the surgery, microbiological results remained negative for the organism. The patient had good lung reexpansion, with full extension into the bony thorax (Fig. 4). With regard to scapular dysfunction, the patient developed slight limitation in movement when raising the left upper limb straight toward the head and putting it on the ear, but there has been no other movement disorder of the upper limb or scapula, and no difficulty in daily life. There is no thoracic deformity or disfigurement.
One month after the decortication, re-infection with Aspergillus occurred and persistent air leak developed. Chest computed tomography revealed parietal pleural thickening in the apical thoracic cavity.