1. Introduction
In 1937, the first case of emphysematous giant bullous emphysema (GBE) was described by Burke as part of the clinical syndrome called “vanishing lung syndrome”. This condition was initially identified in a patient with a history of heavy smoking who presented with large bullae in upper pulmonary lobes, associated with septal emphysema [
1]. Subsequently, radiographic criteria for this pathology were described by Roberts, defining it as the presence of a giant bulla in one or more upper lobes—generally unilateral— occupying at least one-third of the ipsilateral hemithorax and exerting compressive effects on the surrounding lung parenchyma [
2]. GBE may present as isolated or multiple bullae, occurring either as a localized abnormality or in the presence of diffuse lung emphysema. Additionally, this condition can be associated with various risk factors, such as chronic tobacco exposure, and may also occur in patients with genetic conditions like Marfan, Ehlers–Danlos disease and alpha-1 antitrypsin deficiency [
3,
4]. It frequently occurs in patients with chronic obstructive pulmonary disease (COPD) and is associated with a spectrum of clinical manifestations including dyspnea, hypoxia, respiratory failure, and chest pain. However, in some cases, GBE may remain entirely asymptomatic and only be detected incidentally in imaging studies.
High-resolution computed tomography (HRCT) is considered the most reliable and effective diagnostic tool for GBE, as it provides a detailed evaluation of the bullae size, distribution, and the extent of underlying emphysema while also identifying other coexisting lung pathologies [
5].
Surgical resection of these bullae is a well-established treatment and indicated, particularly, in symptomatic patients or those experiencing complications. The primary goal of surgical intervention in GBE is to remove non-functional, hyperinflated lung segments, thereby allowing re-expansion of the compressed, healthier lung parenchyma and improving overall ventilation and gas exchange. The most common complications that may sometimes necessitate surgical intervention include pneumothorax, hemoptysis, progressive respiratory failure, and infection [
6]. Therefore, in certain asymptomatic cases, preventive intervention such as lung volume reduction surgery (LVRS) may be helpful to reduce the risk of such episodes.
Nowadays, several types of surgical approaches have been described, and among these, video-assisted thoracoscopic surgery (VATS) is recognized as a safe and effective surgical technique for LVRS [
7,
8,
9]. Surgical treatment of giant bullae is proven to be effective and associated with low mortality rates [
10]. More recently, Uniportal video-assisted thoracoscopic surgery (U–VATS) has emerged as a refinement of traditional approaches. The first case of a U–VATS giant bullae resection was described in 2016 [
11].
In this study, we present our experience with 29 cases of U–VATS bullectomy for GBE, aiming to evaluate the effectiveness and safety of this technique. By analyzing surgical outcomes, postoperative recovery, and long-term follow up, we seek to further establish U–VATS as a viable approach in in the surgical treatment of GBE.
2. Material and Methods
2.1. Study Design and Patient Selection
This is a retrospective study based on prospectively collected data from two high-volume European thoracic surgery centers between August 2016 and January 2024. This study was approved by the Institutional Review Board (I.R.B.) (approval n°43754/20 Prot. ID.3553) and was therefore performed in accordance with the ethical standards of the Declaration of Helsinki and its later amendments. All patients signed an informed consent before the operation for the anonymous treatment of their clinical data.
Medical records of all patients diagnosed with GBE and referred for surgical treatment following pneumological evaluation were reviewed. The diagnosis was established via clinical presentation and instrumental findings on HRCT scans.
Inclusion Criteria:
Patients diagnosed with GBE confirmed by HRCT.
Patients who underwent Uniportal VATS bullectomy for symptomatic relief or complication management.
Age ≥ 18 years.
Preoperative assessment confirming feasibility for U–VATS surgery.
Availability of complete follow-up data for at least six months after surgery.
Exclusion Criteria:
Patients with diffuse, severe, bilateral emphysema where bullectomy was not feasible.
Patients with significant comorbidities (e.g., severe cardiac dysfunction) contraindicating surgery.
Cases requiring emergency thoracotomy instead of U–VATS.
Incomplete medical records or lack of postoperative follow-up data.
2.2. Preoperative Evaluation
All patients underwent comprehensive preoperative evaluation to assess their surgical eligibility and optimize perioperative management. This included:
Blood analysis: complete blood count (CBC), coagulation profile, serum electrolytes, liver and renal function tests, C-reactive protein (CRP).
Respiratory function tests: spirometry (forced expiratory volume in 1 s [FEV1], forced vital capacity [FVC]), arterial blood gas analysis (pH, pO2, pCO2, HCO3−).
Pneumological evaluation: consultation with a pulmonary specialist to assess functional status, presence of chronic obstructive pulmonary disease (COPD), and smoking history.
Radiological exams: standard chest X-ray for lung pathology screening and HRCT to evaluate bullae size, lung parenchyma involvement, and emphysema severity.
2.3. Surgical Technique
All procedures were performed using a Uniportal VATS approach under general anesthesia with single-lung ventilation. Patients were placed in lateral decubitus position, ensuring optimal thoracoscopic visualization and maneuverability.
A single 3-cm incision at the 5th intercostal space was performed along the middle axillary line, serving as the exclusive access point for both visualization and instrumentation. A wound protector (Alexis
® Small) was used to protect the intercostal space, minimize trauma, and prevent lens contamination of the camera (30°/10 mm) during the procedure. Dedicated long and curved instruments with double articulation, proximal and distal, were used to enhance maneuverability and ensure precise handling of lung tissue within the pleural cavity through the single incision [
12,
13]. The bullectomy was performed (
Figure 1) using staplers with 45- or 60-mm cartridges. In cases of large bullae, especially in patients with severe lung emphysema and fragile pulmonary tissue, where there was a heightened risk of post-operative air-leaks, additional reinforcement of the stapler line was performed. This was achieved by using Gore
® Seamguard
® bioabsorbable reinforcement (
Video S1,
Figure 2), a synthetic buttressing material designed to enhance sealing, reduce staple line dehiscence, and minimize the likelihood of persistent air leaks. Additionally, for patients at high risk of pneumothorax recurrence, selective apical chemical pleurodesis was performed. A sterile talc powder was introduced into the pleural spaces via a catheter and distributed evenly.
At the end of the procedure, a single 28Fr chest drain was positioned through the same Uniportal VATS incision. When pleurodesis was performed, the chest tube was immediately connected to active suction (−20 cm H2O) for at least three days after surgery. The application of negative pressure promoted lung re-expansion, ensuring maximum pleural apposition and enhancing the fibrotic response. In all the other cases, the drainage was maintained under a water seal, with suction applied selectively in cases of incomplete lung re-expansion or postoperative pneumothorax.
Following the U–VATS bullectomy, all patients underwent standardized postoperative monitoring and recovery protocols, ensuring early detection of complications and optimal pulmonary rehabilitation. Postoperatively, all patients underwent serial chest X-ray evaluations and were closely monitored for systematic inflammatory reactions, including transient fever, mild chest discomfort, or pleural effusion, which are common self-limiting effects of talc pleurodesis. The decision to remove the chest drain was guided by both radiological findings and clinical parameters, ensuring adequate lung expansion and controlled fluid drainage with an output consistently ≤ 200 mL over 24 h.
2.4. Statistical Analysis
Continuous variables were expressed as mean ± standard deviation, and categorical variables as percentage. Pearson’s χ2 test and Fisher’s exact test were used to compare categorical variables, and Student’s t-test was used to compare continuous variables.
For all tests, a p-value < 0.05 was considered statistically significant. Statistical analysis was performed using IBM SPSS Statistics for Macintosh (version 25.0, IBM Corp., Armonk, NY, USA).
3. Results
A total of 29 patients diagnosed with GBE underwent U–VATS bullectomy. The preoperative characteristics of the cohort are summarized in
Table 1.
Nineteen out of 29 were males (65.5%), with a mean age of 44.7 ± 8.8 years. In the cohort of 29 patients, 18 (62.0%) presented with a single giant bulla, while the remaining cases involved giant bulla in the context of diffuse pulmonary emphysema. Notably, in 20 patients (68.9%), the bulla was located on the right side. Among these, 19 cases were confined to the upper lobe, while one affected the middle lobe. Left-sided involvement was observed in 31.1% of cases, including one patient presenting a bulla in the left upper lobe with a coexisting non-small cell lung cancer (NSCLC). Radiological assessment revealed that the mean largest bulla diameter, as measured by HRCT scan, was 91.6 mm ± 61.5 mm.
Ten patients (34.5%) were active smokers at the time of surgery. Main comorbidities included COPD in nine patients (31.0%), systemic arterial hypertension in three patients (10.34%), and Basedow disease in one patient (3.4%). The mean preoperative lung functionality tests and arterial blood analysis were as follows: FEV1 100.3 ± 4.1, FVC 96.7 ± 2.4, FEV1/FVC 96.7 ± 2.4, pO2 96 ± 6.4 mmHg, pCO2 35 ± 1.6.
Preoperative symptoms varied among the study cohort, with pneumothorax being reported in four patients (13.8%) before surgery, and one patient requiring chest tube placement. In addition, six patients (20.7%) presented with acute chest pain and dyspnea at the time of admission.
Twenty-eight cases (96.6%) successfully underwent Uniportal video-assisted thoracoscopic (U–VATS) bullectomy without the need for conversion to thoracotomy. Selective apical chemical pleurodesis was performed in 11 cases (37.9%), primarily in those with diffuse emphysema or multiple bullae. In patients with severe emphysema, the stapler line was buttressed with Gore® Seamguard® before performing selective chemical pleurodesis to minimize the risk of postoperative air leaks. Additionally, one patient with a concurrent non-small cell lung cancer (NSCLC) (adenocarcinoma T1bN0M0) underwent a U–VATS left upper lobectomy following an initial wide wedge bullectomy.
The mean operative time was about 74.5 ± 35.1 min, and no conversion to thoracotomy or additional incisions were required. There were no intraoperative complications.
The mean duration of chest tube drainage was 4.5 ± 0.9 days. The mean hospital stay after surgery was 5.6 ± 2.3 days. No major postoperative complications were recorded. However, minor complications occurred in two cases. One patient (3.4%) developed transient atrial fibrillation, which was successfully managed with pharmacological cardioversion. Another patient (3.4%) developed a minimal persistent apical pneumothorax, likely due to inadequate lung re-expansion, without evidence of air leakage, which resolved spontaneously without the need for chest tube reinsertion. Notably, no cases of postoperative air leakage were recorded in this cohort.
Histopathological analysis of the resected specimens confirmed the presence of emphysematous bullae in GBE with a fibrohyaline wall and chronic non-specific inflammatory infiltrates. The mean size of the resected bulla on the specimen was 12.3 ± 3.2 cm (
Figure 3). Long-term follow-up was conducted over a mean time of 22.0 ± 14.0 months. During this period, there were no recurrences of pneumothorax, further confirming the efficacy of U–VATS bullectomy preventing recurrent air leaks and lung collapse.
Pulmonary function tests performed six months postoperatively demonstrated a statistically significant improvement in lung function, especially in FEV 1 values. The mean postoperative FEV1 increased to 107.3 ± 13.8, compared to a preoperative value of 89.3 ± 14.1, (
p = 0.04). Arterial blood gas analysis remained stable with a postoperative pO
2 101 ± 1.1 (
p = 0.126) and pCO
2 35 ± 2.1 (
p = 0.569). The summary of the postoperative results is shown in
Table 2.
4. Discussion
Surgery is considered the treatment of choice for GBE, when feasible, based on clinical conditions. Consequently, LVRS has been demonstrated to facilitate re-expansion of the functional lung parenchyma that is compressed by GBE, thereby improving pulmonary ventilation and gas exchange [
10]. However, this intervention is not suitable for all patients, particularly those with large bullae in a context of extensive, severe emphysema complicated by hypercapnia, where lung resection may further compromise respiratory function rather than improve it. Over the years, many different surgical techniques have been employed in the treatment of bullous emphysema, which are completely abandoned now, including thoracoscopic bullous endoloop ligation, intracavitary bullous drainage, laser bullae ablation, and bullous fibrin glue treatment [
12,
14,
15]. Despite their initial promise and reported outcomes, these methods have been largely abandoned due to suboptimal long-term outcomes and high rates of recurrence. Nowadays, modern surgical approaches emphasize not only survival and mortality rates but also the postoperative quality of life, with a focus on minimizing complications and optimizing lung function recovery.
The introduction of VATS has significantly revolutionized the treatment of pulmonary bullous disease, and now it is the most frequently utilized approach, due to its minimally invasive nature. Indeed, numerous studies have demonstrated that many cases of pulmonary bullous pathologies have been treated successfully using tri-portal and bi-portal VATS [
7,
9], yielding good results in terms of mortality, morbidity, and overall surgical outcomes.
More recently, the refinement of minimally invasive thoracic surgery has led to the introduction of Uniportal VATS, which has been increasingly recognized as a viable alternative for lung volume reduction procedure and bullectomy. A limited number of case reports have described the successful application of the Uniportal VATS approach in the treatment of LVRS [
11,
13,
16]. Lopes et al. described a case of U–VATS bullectomy for vanishing lung syndrome, suggesting that the Uniportal approach reduces surgical trauma, postoperative pain, and the length of hospital stay [
13]. However, the current literature on U–VATS bullectomy in the management of GBE is largely confined to small-scale studies lacking robust long-term follow up data. Existing reports, though limited, suggest positive and encouraging outcomes, particularly in terms of postoperative length of stay, complication rates, and overall feasibility of the surgical technique.
Leveraging our extensive experience with the U–VATS approach in treating various thoracic pathologies, including NSCLC [
17] and esophageal diseases [
18], we aimed to evaluate the effectiveness of this technique in the specific setting of LVRS for large bullae in GBE. U–VATS, for this type of pathology, can be challenging due to the bullae size or their anatomical localization, which can limit visualization and operative maneuverability. As a result, successful execution of the procedure requires an experienced surgeon with refined technical skills in minimally invasive lung surgery.
A potential key advantage of U–VATS over conventional VATS approaches lies in its ability to minimize postoperative pain and neuralgia. This is achieved through the use of a single incision, which reduces trauma to the intercostal nerves compared to the multiple incisions required for bi-portal or tri-portal VATS. Additionally, the application of a wound protector further mitigates intercostal injury during the surgery by preventing direct compression of the nerve and soft tissue, compared to standard trocars commonly used in tri-portal VATS [
19]. These factors contribute to lower levels of postoperative chest pain and discomfort, improving the patient’s recovery by facilitating early ambulation and reducing reliance on opioid analgesia, both of which are particularly beneficial for patients with pre-existing respiratory conditions such as COPD and emphysema.
Furthermore, another major benefit of the Uniportal VATS approach is its excellent exposure of the lung’s anterior and posterior surfaces, which allows for the simultaneous use of multiple instruments (e.g., ring forceps for grasping the lesions, dissectors, energy devices, and staplers), with the enhanced hand–eye coordination akin to an open surgery approach [
20]. The insertion of all instruments through a single incision, working along the same plane (like in a tunnel) may resolve the problem of instrumentation and visualization in a limited space of the pleural cavity due to GBE. This approach minimizes spatial interference between instruments, which can be a significant drawback in other VATS techniques where instruments operate on different planes, leading to difficulties in good lung exposure and instrumentation, and therefore in possible conversions. Despite requiring a high level of surgical proficiency and dexterity in thoracoscopy, our findings suggest that in the hands of experienced surgeons, U–VATS appears to be a safe and feasible method for bullectomy.
Our postoperative results support the efficacy of U–VATS in treating GBE, as demonstrated by improvements in lung function and the absence of major complications. Previous literature reports [
21,
22,
23] suggest that patients undergoing LVRS or bullectomy via VATS may experience significant gains in respiratory function and haemogasanalytic parameters following this surgery, in applicable cases. In our cohort, postoperative pulmonary respiratory function data showed significant improvement in FEV1 values, increasing from 89.3 ± 14.2 to 107.3 ± 13.8 (
p = 0.04) following surgery. Arterial blood gas analysis data remained stable, further supporting the functional benefits of lung volume reduction in these patients.
One of the most concerning postoperative complications following bullectomy and LVRS remains prolonged air leakage. Schipper et al. and Krishnamohan et al. [
21,
22] have noted that up to 53% of patients undergoing VATS bullectomy experienced persistent air leaks, which significantly extended hospital stays and increased morbidity. Additional postoperative complications included atrial fibrillation (12%), the need for postoperative mechanical ventilation (9%), pneumonia (5%), and incisional pain. The risk of prolonged air leakage is particularly relevant in patients with extensive lung emphysema due to the fragile nature of pulmonary tissue, making it more difficult to achieve a secure staple line and to maintain the integrity of suture. Despite these concerns, our findings demonstrated no cases of postoperative air leakage, a finding that stands in contrast to previous reports and highlights the importance of meticulous surgical techniques.
A key factor contributing to this success was the use of selective apical chemical pleurodesis in high-risk cases, which may be crucial to control and prevent air leakage effectively [
24]. Previous studies by Divisi et al. have similarly found that chemical pleurodesis plays a critical role in reducing postoperative air leak duration and improving surgical outcomes [
24,
25]. Our findings reinforce the importance of this adjunctive measure in preventing complications and expediting patient recovery.
Another frequently described complication is pleural empyema due to prolonged air leakage and pleural contamination, particularly in patients with COPD or lung emphysema [
26,
27]. Notably, in our series, we did not observe any cases of air leakage or pleural empyema. This contrasts with existing studies, where air leaks were among the most frequent complications.
In patients with severe emphysema, pulmonary resection was performed using buttressed staplers in conjunction with selective chemical apical pleurodesis to minimize the risk of postoperative air leakage. This approach contributed to shorter hospitalization and faster recovery. In our series, the decision to use buttressed stapler lines or apply chemical pleurodesis was made on a case-by-case basis, particularly in complex cases where emphysematous lung tissue was highly fragile. These measures played a crucial role in ensuring treatment was as radical as possible, stapling on healthy parenchyma rather than on areas of dystrophic or severely damaged tissue, whenever feasible, to minimize the risk of prolonged air leaks. In situations where this was not feasible, the strategies employed in our series may offer significant clinical benefits. However, patient selection for surgery remains a critical determinant of success [
28], especially in cases of severe emphysema, where factors such as age, the presence of comorbidities, baseline respiratory function values, and imaging findings play a pivotal role. Furthermore, an additional consideration is the potential coexistence and occasional occurrence of NSCLC and GBE. The literature reports indicate that lung cancer and emphysematous bullae may exist, with a potential risk of malignancy in large bullae [
29]. In our study, one patient required a left upper lobectomy due to the incidental finding of NSCLC (adenocarcinoma T1bN0M0). This underscores the need for thorough preoperative assessment to ensure appropriate surgical planning and oncological management when necessary.
Limitations and Point of Strength
While this study provides valuable insights into the use of Uniportal VATS (U–VATS) for the treatment of GBE, certain limitations should be acknowledged. First and foremost, it is a retrospective study, which inherently limits the ability to control for potential confounding factors and introduces the risk of selection bias.
Furthermore, the absence of a control group makes it challenging to directly compare U–VATS with other approaches, or with non-surgical management strategies. While the positive outcomes observed in this cohort are encouraging, further comparative studies are necessary to determine whether U–VATS provides superior advantages over alternative treatment options.
Another factor to consider is the sample size. Although this is the largest series of U–VATS bullectomy for GBE reported to date, the number of patients remains relatively small, which limits the statistical power and generalizability of the results. A larger, multi-institutional study with extended follow-up would be beneficial in confirming the long-term effectiveness of this technique.
Despite these limitations, this study represents a significant step forward in assessing U–VATS for GBE, offering valuable clinical data that contribute to the ongoing discussion about optimal surgical management for this condition.
This study has several important strengths that enhance its relevance and reliability. Notably, it is a bicentric study, incorporating data from two high-volume European thoracic surgery centers.
Another major strength is that this study represents the first documented case series of Uniportal VATS bullectomy for GBE with a six-month follow-up, filling a gap in the existing literature. While some previous reports have described isolated cases or small cohorts, our study provides a more structured evaluation of the feasibility and outcomes of this technique in a larger, well-defined patient population.
Lastly, the observed significant improvement in postoperative lung function, particularly in FEV1 values, further supports the clinical benefits of U–VATS for selected patients with GBE. These findings provide a foundation for future prospective studies and help establish U–VATS as a promising minimally invasive approach in the surgical management of GBE.