Surgical management of extensive diaphragmatic and pleural thoracic endometriosis: a case report
Case Report

Surgical management of extensive diaphragmatic and pleural thoracic endometriosis: a case report

Zeuz Islas1, William Carroway2, Jeffrey B. Velotta1,3,4

1Kaiser Permanente Bernard J. Tyson School of Medicine, Kaiser Permanente, Pasadena, CA, USA; 2East Bay General Surgery Department, University of California, San Francisco, San Francisco, CA, USA; 3Department of Thoracic Surgery, Kaiser Permanente Oakland, Oakland, CA, USA; 4Department of Surgery, University of California, San Francisco, San Francisco, CA, USA

Contributions: (I) Conception and design: W Carroway, JB Velotta; (II) Administrative support: JB Velotta; (III) Provision of study materials or patients: JB Velotta; (IV) Collection and assembly of data: Z Islas, W Carroway; (V) Data analysis and interpretation: All authors; (VI) Manuscript writing: All authors; (VII) Final approval of manuscript: All authors.

Correspondence to: Zeuz Islas, BA. Kaiser Permanente Bernard J. Tyson School of Medicine, 98 S Los Robles Ave., Pasadena, CA 91101, USA. Email: zeuz.islas@kp.org.

Background: Thoracic endometriosis is a rare disorder caused by endometriotic tissue infiltration of the diaphragm, pleura, lung parenchyma, and airways, causing significant symptoms and clinical challenges. Diagnosis can be determined, after ruling out other pulmonary diseases, due to the cyclic nature of symptoms that often coincide with the patient’s menstrual cycle. Treatment strategies include hormonal and surgical management, but this can be challenging as it can involve the diaphragm, lung, and chest wall. This case sheds light on the many aspects required for treatment and surgical management. The rare and complex nature of thoracic endometriosis allows this case report to contribute to the limited clinical knowledge of what is currently known. As well as the decision-making process between primary diaphragm repair versus the use of mesh for repair. We present a case that reviews important surgical principles in managing and treating thoracic endometriosis.

Case Description: A 32-year-old female with a history of endometriosis presented to the clinic with catamenial chest and right shoulder pain. Magnetic resonance imaging (MRI) was obtained, confirming the presence of endometriotic lesions in the right posterior diaphragm. The patient’s history included previous laparoscopic pelvic endometriosis surgeries but many of her symptoms persisted. She expressed a desire for pregnancy and opted for surgical excision of the endometriotic lesions. A collaborative surgical effort between obstetrics and gynecology (OBGYN) and thoracic surgery was performed. Right video-assisted thoracoscopic surgery (VATS) diaphragm excision, lung wedge resection, and endometriotic ablation were performed by the thoracic surgery team, followed by laparoscopic ovarian cystectomy and endometrioma resection performed by the OBGYN surgeons. The patient recovered and had vastly improved chest and shoulder pain at her 2 and 12 months follow-up visits.

Conclusions: Surgical management of thoracic endometriosis can be challenging but effective in helping patients with continuous symptoms. This case underscores the importance of multidisciplinary healthcare, patient-centered approaches, and explores the principles of diaphragm repair.

Keywords: Thoracic endometriosis; video-assisted thoracoscopic surgery (VATS); diaphragm repair; multidisciplinary collaboration; case report


Received: 31 October 2023; Accepted: 15 April 2024; Published online: 28 May 2024.

doi: 10.21037/vats-23-69


Video 1 Surgical resection/ablation of all obvious thoracic endometriosis implants and primary diaphragm repair.

Highlight box

Key findings

● Recurrent, refractory pelvic and thoracic endometriosis can be successfully treated with a combined surgical approach involving complete resection of visible endometriosis through video-assisted thoracoscopic surgery.

What is known and what is new?

● Recurrent thoracic endometriosis can be extremely difficult to treat both medically and surgically.

● This report portrays successful collaborative surgical endometriotic removal in a minimally invasive fashion with complete diaphragm resection and primary diaphragm repair.

What is the implication, and what should change now?

● While not guaranteed, the improvement in symptoms after excisional treatment warrant evaluation from a multidisciplinary team of obstetrics and gynecology specialists and thoracic surgeons. Research and data collection regarding outcomes of surgical thoracic endometriosis intervention should continue.


Introduction

Endometriosis is a gynecologic condition defined as “the presence of endometrial-like glands and stroma out of the uterus” (1). This condition typically affects reproductive organs, but can also manifest in atypical locations, including the thoracic cavity (2). Thoracic Endometriosis is a rare disorder caused by infiltration of the diaphragm, pleura, lung parenchyma, and airways with functioning endometriotic tissue, causing significant clinical challenges (3). Diagnosis can be determined, after ruling out other pulmonary diseases, due to the cyclic nature of symptoms that often coincide with the patient’s menstrual cycle (3,4). Symptoms associated with thoracic endometriosis can be extremely debilitating. Patients commonly present with catamenial pneumothorax, catamenial hemothorax, catamenial hemoptysis, lung nodules, chest pain, and pleural effusion (5,6).

Treatment strategies typically include hormonal management with gonadotropin-releasing hormone analogs to regress endometriotic infiltrates, and surgical management, which can be challenging as it can involve the diaphragm, lung, and chest wall (6,7). This case report reviews surgical principles important in managing and treating thoracic endometriosis while exploring common clinical presentations. This report aims to elucidate the clinical presentation, diagnostic work-up and surgical techniques employed to manage thoracic endometriosis, as well as the complicated nature of mesh implementation (8,9). We present this case in accordance with the CARE reporting checklist (available at https://vats.amegroups.com/article/view/10.21037/vats-23-69/rc).


Case presentation

Clinical presentation

A 32-year-old female with a history of pelvic endometriosis presented to the clinic with a new onset of chest and right shoulder pain, which worsened during her menstrual period. The patient had a surgical history of multiple prior laparoscopic excisions of pelvic endometriosis in addition to a laparoscopic ovarian cystectomy. Magnetic resonance imaging (MRI) confirmed the presence of right posterior diagram lesions consistent with endometriosis (Figure 1) and the diagnosis of thoracic endometriosis.

Figure 1 MRI scan showing lesions consistent with endometriosis, as indicated by the circle and arrow on the right posterior aspect of the diaphragm. MRI, magnetic resonance imaging.

The patient trialed hormone therapy and had previous surgical excisions of intraabdominal endometriomas but continued to present with catamenial chest and shoulder pain. Due to the patient’s desire for pregnancy and symptom relief, surgical intervention and excision was agreed upon for treatment.

Thoracic surgery and obstetrics and gynecology (OBGYN) worked in collaboration to perform the patient’s surgery. The thoracic surgery procedure involved right video-assisted thoracoscopic surgery (VATS) diaphragm excision and primary repair right lung wedge resection, endometriotic ablation, while the laparoscopic ovarian cystectomy and endometrioma resection were performed by the OBGYN team. This procedure emphasized the importance of multidisciplinary collaboration.

Following the procedure, an X-ray was obtained (Figure 2) and the patient was discharged on post-operative day 1 (POD 1). Subsequent follow-ups at 2 and 12 months revealed significant improvement to her catamenial chest and shoulder pain.

Figure 2 POD 1 chest X-ray. AP, anteroposterior; POD 1, post-operative day 1.

Surgical technique and procedure

The surgical procedure was planned and executed to address the complex nature of thoracic endometriosis in this patient (Video 1).

Right VATS diaphragm excision

The patient was positioned in lateral decubitus to allow easier access to the thoracic cavity. A small incision was made and allowed entry of the thoracoscope for visualization of the thoracic structures. The surgical team then identified and delineated the lesions on the diaphragm consistent with endometriosis. Full-thickness diaphragm lesions were resected with the use of electrocautery and LigasureTM.

Lung wedge resection

Given the involvement of the lung with endometriotic lesions, lung wedge resections were also performed. The right upper and lower lobes of the lung were targeted, and wedge resections were carried out to remove the affected tissue. This approach aimed to remove the endometriosis while maintaining lung function.

Endometriotic ablation and ovarian cystectomy

The OBGYN team conducted laparoscopic ovarian cystectomy and resections of the endometriomas. The patient’s history of endometriosis required the removal of endometriomas from the ovaries to improve symptoms and improve the potential for future pregnancy.

Argon coagulation and tissue hemostasis

Argon coagulation was used during the procedure to ensure hemostasis and control bleeding. This was especially useful when dealing with delicate structures like the phrenic nerve near the central tendon of the diaphragm and inner liver parenchyma.

Reapproximation and primary repair of the diaphragm

The reapproximation of the large defect in the diaphragm was performed with attention to size and tension. The edges of the defect were brought together with 2-0 vicryl running V-lock sutures to ensure a tension-free closure. This was a crucial step to warrant proper healing and function of the diaphragm. A tension-free closure is important to avoid complications like a diaphragmatic hernia, which can lead to respiratory issues or visceral ischemia. Primary diaphragm repair is feasible even in large diaphragm defects.

Strattice mesh implementation

The option of mesh placement can also be considered when primary repair alone might not provide sufficient support or when the defect is too large. The use of a Strattice mesh was previously used in many different cases at our institution and provided good results. The mesh, which is composed of porcine acellular dermal matrix, provides structural reinforcement and support to the repaired diaphragm. Due to the minimal tension on a thin diaphragm and the location of the defect directly over the dome of the liver, primary repair was favored over the use of a mesh.

Multidisciplinary collaboration

Collaboration between the thoracic surgery and OBGYN teams was required for the success of the operation. Effective communication and coordination were pivotal during the procedure. This approach emphasizes the importance of a holistic approach to patient care that considers both the gynecologic and thoracic aspects of thoracic endometriosis.

Postoperative care and follow-up

The patient was closely monitored, and an X-ray was obtained (Figure 2) following the procedure in the post-operative period. The patient was discharged on POD 1. Subsequent follow-ups at 2 and 12 months post-operative revealed significant improvement in the patient’s catamenial chest pain, indicating a successful surgery. This reaffirmed the success of surgical intervention in managing the patient’s symptoms and endometriosis.

All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for publication of this case report and accompanying images and video. A copy of the written consent is available for review by the editorial office of this journal.


Discussion

This case explored thoracic endometriosis and its diagnosis, while also investigating the principles of a repair and highlighting the choice between primary-based and mesh-based repair. Regarding diagnosis and more specifically, diagnostic imaging, the role of an MRI versus computed tomography (CT) for endometriotic thoracic implants can be further explored.

CT imaging offers the ability to rule out other diagnoses and provides mapping for surgical planning with high spatial resolution. However, it does have a few drawbacks, including limited contrast resolution and radiation exposure. MRI, on the other hand, offers superior contrast resolution, which is useful in identifying hemorrhagic lesions and diaphragmatic or pleural implants without radiation exposure. MRI imaging tends to have a lower spatial resolution compared to CT (4). The choice between the two depends on diagnostic needs, with MRI being preferable for pleural implants and lesions, making it the superior choice for our case.

Concerning the choice between primary and mesh-based repair, many factors played a role in the decision including the size and location of the defect to ensure that the closure was tension-free. Different mesh options were discussed, including the use of synthetic and biological meshes. Previous use and experience with the Strattice mesh have shown good results (8,9).

The diaphragm repair began with resection of the full-thickness diaphragm lesion. Resection is preferred over ablative methods given full-thickness endometriotic implants are typically associated with fenestrations. Resection can improve the recurrence rate and allows for pathologic evaluation. The diaphragm repair provided a tension-free approach which is crucial in avoiding future complications such as a diaphragmatic hernia (10). This was followed by Argon coagulation of the lesion adjacent to the inferior vena cava (IVC) and right phrenic nerve. Afterwards, wedge resections of the right upper and lower lobes of the lung were performed, along with Argon coagulation of the liver capsule. The procedure finished with the re-approximation and primary repair of the diaphragm defect. Defects over the right diaphragm have a protective effect from abdominal herniation due to the position of the liver.

Recent research on patients with diaphragmatic lesions highly recommends VATS to explore the thoracic cavity. This helps ensure complete identification and excision of endometriotic infiltrates in the thoracic cavity and of the diaphragm. Direct inspection offers the most precise detection, as abdominal laparoscopic management of diaphragm lesions alone seems to be associated with high recurrence rates (11).

This surgical procedure demonstrated successful minimally invasive surgical management of thoracic endometriosis through direct primary repair.


Conclusions

This case spotlights the intricacies of surgical procedures for treating thoracic endometriosis and emphasizes the importance of collaboration among different specialties. The successful combination of right VATS diaphragm excision and primary repair, lung wedge resection, endometriotic ablation, and ovarian cystectomy demonstrated the potential benefits of surgical intervention in cases of refractory thoracic endometriosis. Moreover, the principles of diaphragm repair and consideration of mesh usage provide further insight into the surgical decision-making process, ensuring the best possible outcome for the patient’s well-being and potential future pregnancy.


Acknowledgments

Funding: None.


Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://vats.amegroups.com/article/view/10.21037/vats-23-69/rc

Peer Review File: Available at https://vats.amegroups.com/article/view/10.21037/vats-23-69/prf

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://vats.amegroups.com/article/view/10.21037/vats-23-69/coif). J.B.V. serves as an unpaid editorial board member of Video-Assisted Thoracic Surgery from September 2023 to August 2025. 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. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for publication of this case report and accompanying images and video. A copy of the written consent is available for review by the editorial office of this journal.

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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doi: 10.21037/vats-23-69
Cite this article as: Islas Z, Carroway W, Velotta JB. Surgical management of extensive diaphragmatic and pleural thoracic endometriosis: a case report. Video-assist Thorac Surg 2024;9:33.

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