A new surgical technique of uniportal video-assisted thoracic surgery for thoracic endometriosis-related pneumothorax considering the extent of thoracic endometriosis lesions

Catamenial pneumothorax was first reported by Maurer et al. in 1958 (1) involving a patient with a right spontaneous pneumothorax and endometrial tissue histopathologically confirmed in the diaphragm. Since then, catamenial pneumothorax has been defined as a recurrent, spontaneous pneumothorax occurring during menstruation in women of reproductive age (2), but in recent years, the definition has been questioned because pneumothorax caused by endometrial tissue in the pleural cavity during non-menstruation has also been reported, as well as primary spontaneous pneumothorax (PSP) during menstruation (3-5). Several recent reports have defined catamenial pneumothorax more strictly as thoracic endometriosis-related pneumothorax (TERP) to better reflect the essential pathophysiology (6,7). The pathogenesis of TERP has not been fully investigated, but there are theories that endometrial tissue grows on the diaphragm, reaches the thoracic cavity via the diaphragm, and is caused by visceral pleural endometrial tissue failure or air entering through diaphragm fenestrations (1,8). Indeed, endometrial tissue has been identified in the visceral and parietal pleura or diaphragm and these pleural and diaphragmatic lesions are thought to be linked to the development of a pneumothorax. Surgery is currently considered an effective treatment option for TERP (8,9). Although the thoracoscopic approach has been reported to be useful in recent years, the postoperative recurrence rate remains high (7,9,10). Therefore, various treatment strategies have been reported based on the localization of endometrial tissue in the thorax, as well as the addition of postoperative hormonal therapy (10,11). In recent years, improvements in surgical techniques and the evolution of surgical devices have led to the development of uniportal video-assisted thoracic surgery (VATS) as a less invasive surgical technique compared to multi-portal VATS and robot-assisted thoracic surgery. Beginning with the first report of uniportal VATS lobectomy for lung cancer in 2010, a large number of uniportal VATS procedures have been performed to date and non-inferiority to other surgical techniques with respect to complications and cancer outcomes has been reported (12,13). Specifically, uniportal VATS is considered a more adequate approach for partial lung resection and surgery for PSP. We selected uniportal VATS as a first-line approach for PSP surgical cases. Three-port VATS has been the basic approach for TERP due to the complexity of the surgical technique, which includes diaphragmatic resection plus reconstruction with sutures, partial lung resection, resection and cautery of dorsal parietal pleural lesions, and covering of the visceral and parietal pleura. However, our proficiency with uniportal VATS has led us to attempt to introduce this approach for more difficult pneumothorax cases (14,15). Furthermore, the introduction of uniportal surgery in this procedure offers the advantage of performing surgical techniques coaxially, particularly during diaphragm resection and reconstruction. Considering that the treatment targets young women, we determined it would also be beneficial from an aesthetic perspective. On the other hand, while no clear exclusion criteria have been established for uniportal VATS as an adaptation to TERP, cases requiring reoperation should consider total pleural covering reported by Kurihara et al. (16). Additionally, if significant intrathoracic adhesions are observed during the initiation of uniportal VATS, transition to multi-port VATS should also be considered. We introduced the uniportal VATS technique for TERP in our institution and have successfully managed several cases. This manuscript clarifies the detailed technique of less invasive uniportal VATS for TERP and reports the outcomes of recent cases. Surgical strategies for TERP VATS surgery was selected for diagnostic and therapeutic purposes in patients with TERP. There is no consensus on the extent of the procedure, which can vary from resection and suturing of the diaphragm (the most common finding) to resection of the pulmonary apex and ablation of the pleural endometrial lesion. A high recurrence rate of 20–40% has been reported with these procedures and some reports suggest that postoperative pleurodesis and hormonal therapy may reduce the recurrence rate (9,11,17). Ochi et al. (18) reported on localization of thoracic endometrial lesions in 160 patients who underwent VATS for TERP. All patients had diaphragmatic lesions. Visceral pleural lesions were most common in S4, followed by dorsal lesions from S4 (S2 and S6), which accounted for 82.7% of visceral pleural lesions. Parietal pleural lesions were most common in the dorsal area of the 6th intercostal space, followed by the upper and lower intercostal spaces, with 86.1% of pleural wall lesions in the dorsal areas of the 4th–9th intercostal spaces. In addition, it was reported that multi-port VATS pleural covering with oxidized regenerative cellulose (ORC) mesh reduced the 2-year postoperative recurrence rate to approximately 19.3% (18). Based on this report, the basic surgical technique for TERP in our institution includes the following: partial resection and reconstruction of the diaphragm with visible endometrial tissue; partial resection of the lung with visceral pleural findings centered on S4; and coverage with ORC mesh over the S2, S4, and S6 regions and the 6th dorsal intercostal space (Figure 1). Dual covering with polyglycolic acid (PGA) sheets under the ORC mesh is also used for diaphragmatic reconstruction and partial lung resection. Additionally, this surgical procedure is expected to utilize multiple ORC sheets (6 to 7 sheets). This approach references and applies the total pleural covering technique reported by Kurihara et al. as an effective procedure for refractory pneumothorax (16,19). We present this article in accordance with the SUPER reporting checklist (available at https://vats.amegroups.com/article/view/10.21037/vats-25-22/rc). Case presentation The patient was a 40-year-old woman who had right chest pain associated with her menstrual cycle for approximately 6 months prior to seeking evaluation at our institution. Three months before seeking evaluation at our institution, she became aware of right chest pain and dyspnea that increased with her menstrual cycle. She was referred to a general hospital by a local physician, where a chest X-ray showed severe right lung collapse. She underwent right thoracic drainage with a diagnosis of a complete lung collapse. Endometriosis-related pneumothorax was suspected, given the right-sided chest pain associated with previous menstrual cycles. The patient was treated conservatively with thoracic drainage and was referred to our institution for surgery due to desired fertility and concerns about the increased risk of an endometriosis-related pneumothorax recurrence with fertility treatment. The patient considered the timing of surgery, but 3 months later, she had another episode of right chest pain and sought emergency evaluation in our institution, where she was diagnosed with a right complete lung collapse (Figure 2). She was admitted after right thoracic drainage. Because the patient had a recurring complete lung collapse, the decision was made to continue hospitalization and perform surgery. A uniportal VATS partial diaphragm resection plus reconstruction, partial lung resection, resection and cauterization of dorsal parietal pleural endometriotic lesions, and pleural covering procedure was performed under general anesthesia and one-lung ventilation to resolve the pneumothorax and diagnose thoracic endometriosis. The surgical procedures and other relevant details are described in the following paragraphs. Preoperative preparation and requirements On the day of admission, the patient was managed with a water seal and the following day, continuous suctioning was performed at −7hPa. The air leak was resolved by hospital day 3. An electrocardiogram showed no abnormalities and blood tests were normal, so the patient was considered a good surgical candidate. The surgery was performed by two thoracic surgeons, a resident, an anesthesiologist, and two operating room nurses. The patient was fixed in the left lateral decubitus position under general anesthesia and a pillow was placed in the left axilla so that the right intercostal space was as wide as possible. The surgical field was disinfected with iodine and a cephazolin infusion was administered at the time of surgery according to Centers for Disease Control and Prevention guidelines. This study was conducted in accordance with the Declaration of Helsinki and its subsequent amendments. The study was approved by the Ethics Review Committee of Chibaken Saiseikai Narashino Hospital (No. 2025-12). Regarding the handling of videos and clinical data related to this paper, consent was obtained through an opt-out procedure under the hospital’s internal ethics review. The information disclosure document for this study can be accessed on the hospital’s website. Step-by-step description Surgical procedure An incision approximately 3 cm in length was made above the anterior axillary line in the 6th intercostal space (Figure 3). The subcutaneous tissue and muscle layer were dissected to the right thoracic cavity. An S-size Alexis® O Wound Protector/Retractor (Applied Medical, Rancho Santa Margarita, CA, USA) was used to assist with the open chest. The entire thoracic cavity was observed in detail thoracoscopically. In addition to the diaphragm, the visceral pleura at the so-called tri-lobar confluence from S4 to S2 and S6 and the dorsal parietal pleura of the 4th–9th intercostal spaces are frequently involved in patients with TERP, so observations focused on these lesions. It was also essential to observe the apex of the upper lobe of the lung, which is the preferred site of pulmonary cysts in PSP. In this patient, we observed numerous fenestration lesions in the diaphragmatic tendon center. In addition, a visceral pleural lesion with bullous changes was identified at the interlobar region of S4. In addition, a similar visceral pleural lesion was identified at S7/S8. A pleural lesion with multiple vascular growths was identified approximately 1 cm above the sympathetic trunk between the 6th and 10th intercostal spaces in the dorsal parietal pleura. No obvious pulmonary cysts were observed in the apex of the upper lobe (Video 1). Diagnostic and therapeutic partial lung resections were performed on the S4 and S7/S8 visceral pleural lesions with an Endo GIA™ 45 mm Articulating Medium/Thick Reload with Tri-Staple™ Technology (Medtronic, Minneapolis, MN, USA). The dorsal parietal pleural lesions were subsequently cauterized with ball-type electrocautery (Video 1). Next, a partial resection and reconstruction of the diaphragm was performed. The inferior vena cava was identified and the border between the diaphragmatic tendon center and the muscle layer approximately 2 cm lateral to the vein was cauterized with a LigaSure™ Maryland (Medtronic) to create a fistula between the thoracic and abdominal cavities. A diaphragmatic dissection was performed with an automated suture machine to fully contain fenestration lesions from the same site. The tendon center was treated with an Endo GIA™ 45 mm Articulating Vascular Medium Reload with Tri-Staple™ Technology (Medtronic) and the muscle layer was treated with an Endo GIA™ 45 mm Articulating Medium/Thick Reload with Tri-Staple™ Technology. The fistula was single-knot sutured with a total of 12 stitches using 2-0 coated Vicryl (Ethicon, Raritan, NJ, USA) so that the staple line was aligned (Video 2). The chest cavity was flushed with sterile distilled water and a water sealing test was performed to confirm the absence of a pulmonary air-leakage before proceeding to covering. First, the apex of the lung was covered with ORC mesh. This lesion is a frequently occurring lesion of pulmonary cyst for PSP. Then, the interlobar area was covered. In the area where partial resection was performed, covering with a PGA sheet was performed, followed by covering with ORC mesh. The dorsal visceral pleura from the center of the interlobar and the dorsal parietal pleura, where thoracic endometrial tissue is often observed, were covered with ORC mesh. The mesh was applied in order from the ventral side. It is important to occasionally apply fibrin glue to strengthen the attachment to the lungs and to prevent the mesh from migrating. Finally, a PGA sheet and ORC mesh were used to cover the reconstructed diaphragm and fibrin glue was spread over the entire area (Video 3). The extent of lung expansion and pleural coverage was checked. After confirming hemostasis and performing an intercostal nerve block with ropivacaine, a thoracic drainage tube was inserted into the chest cavity through the same incision. Postoperative considerations and tasks There is often a cloudy pleural effusion due to chemical pleural stimulation and dissolution of the sheet ingredients when multiple ORC mesh sheets are used. Therefore, it is recommended that a thicker drainage tube be used for detention. In this case, an 8 mm thoracic drain was used. Postoperative management of the thoracic drainage tube was routinely performed as follows: (I) continuous low-pressure suction (−7hPa); (II) continuous periodic milking of the thoracic drain; and (III) placement of the drain until the daily drainage volume was <200 mL. The drain was removed on postoperative day 3 and the patient was discharged home on postoperative day 6 with no Clavien-Dindo grade ≥3 complications. The postoperative histopathologic evaluation revealed a diagnosis of TERP (Figure 4). Tips and pearls - During surgery for TERP, the distribution of endometrial lesions in the thoracic cavity should be carefully considered. - In particular, the treatment of diaphragmatic lesions as well as the covering of S2, S4, and S6, and the observation, cautery treatment, and covering of the dorsal wall pleura may lead to a lower postoperative pneumothorax recurrence rate. - The uniportal VATS approach with a surgical wound of approximately 3 cm at the 6th intercostal anterior axillary line is sufficient to perform these procedures. Proper technique and coordination with the camera assistant are important. - The diaphragm suture requires looking down in the multi-port VATS, whereas the needle can be moved coaxially with the visual field in uniportal VATS, making the procedure easier.

In a review article, Marjański et al. (20) reported that the VATS approach is basically used in surgery for TERP, with bullectomy, pleurectomy, and pleurodesis as techniques, and diaphragmatic resection is performed when there are lesions involving the diaphragm. If there is extensive diaphragmatic involvement, a thoracoscopically assisted small thoracotomy is used. Although there may be differences in the approach between institutions, a multi-portal VATS has been conventionally performed at our institution. We have used a 3-port VATS approach in which a camera port is placed on the 6th intercostal posterior axillary line for port placement and manipulation ports are placed on the 8th intercostal anterior and 9th intercostal posterior axillary lines to process the diaphragm. The camera port is changed each time. Using this approach, the diaphragm is assessed by looking down through a thoracoscope during the procedure; hand-eye coordination may be difficult due to rotation of the camera during field of view development. In addition, it is necessary to hold the needle back in the thoracic cavity when suturing the diaphragm and it is difficult to adjust the insertion angle of the automatic suture device and the needle insertion angle. In the case of uniportal VATS, as shown in Video 2, the field of view and the automatic suture device and needle holder are almost coaxial, which makes the procedure easy to perform. In contrast, interference between thoracoscopy and instruments is an important problem in uniportal VATS. The training required for this surgical technique may include the following. First, sufficient proficiency in intrathoracic manipulation during uniportal VATS is essential. To achieve safer and more efficient uniportal VATS procedures, it is essential to facilitate collaboration between surgeons and endoscopists by learning appropriate instrument selection and insertion techniques through uniportal VATS procedures such as partial lung resection and PSP surgery. Second, regarding diaphragm resection plus reconstruction, proficiency in needle manipulation during multi-port VATS is essential. The following points should be noted. It should be noted that there is a risk of injury due to interference of the jaws of the automatic suture device with the hepatic right triangular ligament or hepatic parenchyma during insertion of the suture device during diaphragmatic resection. Furthermore, due to the extensive coverage of the visceral pleura, concerns exist regarding postoperative pulmonary atelectasis and restrictive dysfunction. Therefore, adequate postoperative drainage is essential. While a surgery for PSP typically allows for approximately one day of postoperative drainage, this surgical technique necessitates a relatively longer drainage period of about three days. Finally, regarding medical costs, since multiple ORC sheets (5–7 sheets) are usually needed, in Japan’s insurance-based medical care system, approximately half of the costs of ORC sheets are cut during insurance reviews, and the excess amount must be covered by the hospital. We have performed six cases with uniportal VATS for TERP from November 2023 to the present. Clinical data for each case are shown in Table 1. The operative time was 162.5±9.7 min, the blood loss was 68.3±132.1 mL, the drainage period was 3.1±0.4 days, and the time-to-discharge was 5.6±0.5 days. Pathological diagnosis of endometrial tissue within the thoracic cavity was confirmed in 5 of 6 cases. In one case, although histological diagnosis was not possible, a distinct diaphragmatic lesion was identified intraoperatively, leading to a clinical diagnosis of TERP. Additionally, postoperative hormonal therapy was initiated in 2 of 6 cases. There were no Clavien-Dindo grade >3 complications and no postoperative recurrences. A comparison of the incidence of pneumothoraxes before and after surgery within a given time period is shown in Figure 5. For reference, we show the clinical data of multi-port VATS performed in our department in Table 2. Considering the learning curve for the surgical technique, we selected the most recent six cases. One case of postoperative recurrence was observed. The operative time tended to be longer at 223.8±16.9 min. Furthermore, the postoperative drainage period and hospital stay also tended to be longer at 6.0±3.72 and 10.0±2.96 days, respectively. To compare TERP surgical outcomes with those of other institutions, we referred to several articles and showed them in Table 3 (17,21-24). While significant differences in treatment strategies and surgical techniques for TERP make accurate comparisons difficult, recurrence rates after surgery have been reported to be around 30%. Therefore, we consider our uniportal VATS technique for TERP may be effective in controlling TERP. However, it should be noted that this study is a retrospective review of a small case series (6 cases) with a short observation period, and therefore the data are preliminary. Further studies with longer-term, prospective follow-up are considered necessary. Table 1 | Case | Age, years | Operation time, min | Bleeding, g | Postoperative drainage period, days | Postoperative hospitalization period, days | Complication (Clavien-Dindo grade >3) | Pathological diagnosis | Postoperative hormonal therapy | Postoperative observation period, days | Recurrence | |---|---|---|---|---|---|---|---|---|---|---| | 1 | 35 | 159 | 0 | 3 | 6 | None | Diaphragm, lung | None | 343 | No | | 2 | 31 | 174 | 0 | 3 | 6 | None | Diaphragm | None | 617 | No | | 3 | 30 | 168 | 0 | 3 | 5 | None | None (intraoperative macroscopic diaphragmatic lesions) | None | 188 | No | | 4 | 43 | 158 | 80 | 3 | 6 | None | Diaphragm, lung | Dienogest | 367 | No | | 5 | 49 | 169 | 0 | 4 | 6 | None | Diaphragm | None | 210 | No | | 6 | 38 | 147 | 330 | 3 | 5 | None | Diaphragm | Dienogest | 139 | No | | Average | 38 | 162.5±9.7 | 68.3±132.1 | 3.1±0.4 | 5.6±0.5 | – | – | – | 346.8±202.9 | – | Data was presented as average ± standard deviation or as individual values, unless otherwise specified. TERP, thoracic endometriosis-related pneumothorax; VATS, video-assisted thoracic surgery. Table 2 | Case | Age, years | Operation time, min | Bleeding, g | Postoperative drainage period, days | Postoperative hospitalization period, days | Complications (Clavien-Dindo grade ≥3) | Pathological diagnosis | Postoperative hormonal therapy | Postoperative observation period, days | Recurrences | |---|---|---|---|---|---|---|---|---|---|---| | 1 | 38 | 225 | 30 | 6 | 8 | None | Diaphragm | Dienogest | 492 | Yes† | | 2 | 45 | 246 | 0 | 4 | 11 | None | Diaphragm | Dienogest | 873 | No | | 3 | 41 | 205 | 0 | 8 | 8 | None | Diaphragm | Dienogest | 797 | No | | 4 | 41 | 206 | 0 | 3 | 7 | None | Diaphragm, lung | None | 386 | No | | 5 | 30 | 240 | 70 | 13 | 15 | None | Diaphragm | None | 748 | No | | 6 | 37 | 221 | 0 | 4 | 11 | None | Diaphragm, lung | None | 699 | No | | Average | 38.6 | 223.8±16.9 | 16.6±28.7 | 6.0±3.72 | 10.0±2.96 | – | – | – | 666±187.8 | – | Data was presented as average ± standard deviation or as individual values, unless otherwise specified. †, minor lung collapse requiring no drainage at 3 months postoperatively. TERP, thoracic endometriosis-related pneumothorax; VATS, video-assisted thoracic surgery. Table 3 | Study | Number of patients | VATS cases [number of uniportal] | With hormonal therapy | Recurrence rate | |---|---|---|---|---| | Andres et al. 2020 (17) | NA | 443 [NA] | NA | 29.0% | | Campisi et al. 2022 (21) | 16 | 15 | 16 | 12.5% | | Kim et al. 2024 (22) | 41 | 41 | 27 | 29% | | Nikolettos et al. 2024 (23) | 220 | NA | 220 | 26.8% | | Gatteschi et al. 2025 (24) | 36 | 34 [6] | NA | 41.7% | | Ito et al. 2025 | 6 | 6 [6] | 3 | 0 | NA, not available; TERP, thoracic endometriosis-related pneumothorax; VATS, video-assisted thoracic surgery.

In this article, we described in detail the surgical technique and treatment strategy of uniportal VATS for TERP. We consider the uniportal VATS approach to be potentially effective in terms of outcomes and complications as a treatment strategy that considers the localization of endometrial tissue in the treatment of frequently recurrent TERP. We hope that our experience with this technique will contribute to the treatment strategies for TERP. Acknowledgments We thank JAM Post Biomedical Communications (Seattle, Washington, USA) for their excellent English language editing service. Footnote Reporting Checklist: The authors have completed the SUPER reporting checklist. Available at https://vats.amegroups.com/article/view/10.21037/vats-25-22/rc Peer Review File: Available at https://vats.amegroups.com/article/view/10.21037/vats-25-22/prf Funding: None. Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://vats.amegroups.com/article/view/10.21037/vats-25-22/coif). T.M. serves as an unpaid editorial board member of Video-assisted Thoracic Surgery from August 2025 to July 2027. The other authors have no conflicts of interest to declare. Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. The study was conducted in accordance with the Declaration of Helsinki and its subsequent amendments. The study was approved by the Ethics Review Committee of Chibaken Saiseikai Narashino Hospital (No. 2025-12). Regarding the handling of videos and clinical data related to this paper, consent was obtained through an opt-out procedure under the hospital’s internal ethics review. The information disclosure document for this study can be accessed on the hospital’s website. Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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