Thoracoscopic treatment of recurrent catamenial pneumothorax

Catamenial pneumothorax is a rare but potentially life-threatening condition that affects women of reproductive age. It is characterized by the recurrent occurrence of pneumothorax during or around the menstrual period. The pathophysiology of catamenial pneumothorax is not well understood, but is thought to be related to the presence of endometrial tissue in the pleural cavity. Case Presentation: A 42-year-old woman was referred to our outpatient department for further thoracic surgical treatment. She already had two previous episodes of pneumothorax that occurred during her menstrual period. A chest X-ray revealed a right-sided pneumothorax and she was admitted to the hospital where a chest tube was placed to treat the pneumothorax. CT scan revealed the presence of endometrial tissue under the right diaphragm. The patient underwent a total laparoscopic hysterectomy to remove the source of endometrial tissue. The subdiaphragmatic endometrial tissue was also removed. She subsequently underwent thoracoscopic treatment to treat the pneumothorax.

The aim of this study was to review the clinical features, diagnostic criteria, and treatment options for catamenial pneumothorax. In addition, the study highlights the importance of a multidisciplinary approach to the management of catamenial pneumothorax, involving pulmonologists, gynecologists, and thoracic surgeons. Further studies are needed to optimize the management of this rare condition.

Catamenial pneumothorax is a rare but potentially life-threatening condition that affects women of reproductive age. It is characterized by the recurrent occurrence of pneumothorax during or around the menstrual period. The pathophysiology of catamenial pneumothorax is not well understood, but is thought to be related to the presence of endometrial tissue in the pleural cavity. Case Presentation: A 42-year-old woman was referred to our outpatient department for further thoracic surgical treatment. She already had two previous episodes of pneumothorax that occurred during her menstrual period. A chest X-ray revealed a right-sided pneumothorax and she was admitted to the hospital where a chest tube was placed to treat the pneumothorax. CT scan revealed the presence of endometrial tissue under the right diaphragm. The patient underwent a total laparoscopic hysterectomy to remove the source of endometrial tissue. The subdiaphragmatic endometrial tissue was also removed. She subsequently underwent thoracoscopic treatment to treat the pneumothorax.

The aim of this study was to review the clinical features, diagnostic criteria, and treatment options for catamenial pneumothorax. In addition, the study highlights the importance of a multidisciplinary approach to the management of catamenial pneumothorax, involving pulmonologists, gynecologists, and thoracic surgeons. Further studies are needed to optimize the management of this rare condition.

Catamenial pneumothorax is a rare but potentially life-threatening condition that affects women of reproductive age. It is characterized by the recurrent occurrence of pneumothorax during or around the menstrual period.1 The pathophysiology of catamenial pneumothorax is not well understood, but is thought to be related to the presence of endometrial tissue in the pleural cavity.2 Here, we present the case of a 42-year-old woman with recurrent catamenial pneumothorax who underwent a hysterectomy and subsequent thoracoscopic treatment. This article also aims to review the clinical features, diagnostic criteria, and treatment options for catamenial pneumothorax. CASE PRESENTATION A 42-year-old woman was referred to our outpatient department for further thoracic surgical treatment. She had two previous episodes of pneumothorax that occurred during her menstrual period. On physical examination, she had decreased breath sounds on the right side of her chest on both occasions. A chest X-ray revealed a right-sided pneumothorax, and she was admitted to the hospital where a chest tube was placed to treat the pneumothorax (Figure 1). Further investigations, including a computed tomography (CT) scan, had already been performed at another hospital abroad to identify the underlying cause of the recurrent pneumothorax. The previously performed CT scan had revealed the presence of endometrial tissue under the right diaphragm. The patient had undergone a total laparoscopic hysterectomy two months ago to remove the source of endometrial tissue. The subdiaphragmatic endometrial tissue had also been removed. She subsequently underwent thoracoscopic treatment to treat the pneumothorax. Due to the patient's young age and limited visibility of endometrial tissue on CT scans, a preoperative CT scan was not performed, and the decision was made to proceed directly to thoracoscopy, as a preoperative CT scan would not change the therapeutic approach. Thoracoscopy did not identify endometrial tissue in the pleural cavity. Two holes in the diaphragm were identified and sutured endoscopically with 4-0 polypropylene sutures (Figure 2). A total thoracoscopic pleurectomy was also performed (Figure 3). The postoperative course was uneventful, and the patient remained stable throughout her hospital stay and was discharged home on oral contraceptives and close follow-up by her pulmonologist and gynecologist (Figure 4). Histology showed no signs of endometriosis. Follow-up examinations by the pulmonologist revealed no further episodes of pneumothorax.

Catamenial pneumothorax is a rare medical condition that accounts for approximately 3–6% of all cases of spontaneous pneumothorax.3 Women of reproductive age are primarily affected, with an average onset age of 35 years.2 Catamenial pneumothorax can be the first manifestation of the thoracic endometriosis syndrome (TES), and is attributed to the presence of endometriotic lesions in the pulmonary and pleural tissues.4 Endometriosis, a prevalent clinical disease among women, usually occurs in the pelvic region but in rare cases can also manifest in extrapelvic regions including the abdominal wall, vulva, perineum, liver, skin, central nervous system, and thoracic cavity.5 The clinical manifestations of thoracic endometriosis include catamenial pneumothorax, hemothorax, and hemoptysis. Patients with these manifestations of spontaneous pneumothorax are often diagnosed without prior comprehensive gynecological evaluation to exclude the existence of catamenial pneumothorax.6 The pathophysiology of catamenial pneumothorax remains a subject of limited understanding.7 Various theories concerning catamenial pneumothorax have been postulated.8 The main theories include the following:2 • Physiological hypothesis. • Metastatic or lymphovascular microembolization theory. • Transgenital–transdiaphragmatic passage of air theory. • Migration theory. According to the “physiological hypothesis”, prostaglandin F2 levels increased during menstruation can trigger vasoconstriction and bronchospasm, leading to alveolar rupture and consequently pneumothorax.2 According to the “metastatic or lymphovascular microembolization theory”, endometrial tissue is transported to the lungs via the venous or lymphatic system. Subsequently, periodic menstruation-induced necrosis of endometriotic lesions adjacent to the visceral pleura results in pneumothorax. In certain cases, hemoptysis can also occur when the endometriotic foci are centrally located.2 The “transgenital–transdiaphragmatic passage of air theory” suggests that atmospheric air travels from the vagina to the uterus, then through the fallopian tubes into the peritoneal cavity, and finally enters the thoracic cavity via diaphragmatic defects.9 According to the “migration theory”, endometrial cells migrate from the lower pelvic region through the peritoneal circulation, eventually reaching subdiaphragmatic areas, predominantly on the right side. This is attributed to the clockwise circulation of peritoneal fluid. Periodic necrosis of endometrial tissue leads to diaphragmatic penetration, resulting in the entry of endometrial tissue into the thoracic cavity. This endometrial tissue implants itself onto the visceral pleura and causes alveolar rupture through cyclical necrosis, leading to pneumothorax.2 Symptoms of catamenial pneumothorax typically manifest around the menstrual period (from 24 hours before to 72 hours after the onset of menstrual bleeding), but they can also occur at other times. Catamenial pneumothorax typically presents with an abrupt onset of chest pain and dyspnea.1,2 A common symptom is pain associated with the menstrual cycle, often localized on the right side of the chest, in the scapular or shoulder area. This localization can be attributed to the shared C5 spinal segment innervation between the diaphragm and these regions. Pneumothorax mostly occurs on the right side, with less frequent occurrences on the left side or bilaterally. The exact reason for this preference is not yet fully understood. Patients may also experience coughing and fatigue.1 The diagnosis of catamenial pneumothorax is based on the patient's clinical history, physical examination findings, and imaging studies. Various imaging modalities such as chest X-ray, CT scans, or magnetic resonance imaging (MRI) are used to confirm the diagnosis.4 The definitive diagnostic criterion is evidence of the presence of endometrial tissue in the pleural cavity, typically by biopsy or thoracoscopy1 While MRI appears to be more accurate in detecting endometriotic lesions in the abdomen, it is also used as a diagnostic tool to identify endometriotic lesions associated with TES. It exhibits particular precision in detecting endometriotic lesions in the diaphragmatic and pericardial regions, but is less accurate in diagnosing visceral, parietal pleural, or bronchopulmonary endometriosis.4 Treatment for catamenial pneumothorax depends on the severity of the condition. Small pneumothoraxes may resolve spontaneously, but larger ones require intervention. Acute pneumothorax can be treated by thoracentesis or chest tube placement. For recurrent or persistent pneumothorax, surgical interventions such as video-assisted thoracic surgery (VATS) or thoracotomy may be warranted. VATS is typically the preferred treatment, while thoracotomy remains an option, especially in reoperations.2 Surgery is ideally performed during the menstrual period, as symptoms and pathological findings are more conspicuous.7 Intraoperatively, endometrial implants were observed in 59.3% of patients, and histopathology confirmed the presence of thoracic endometriosis in 52.3% of patients diagnosed with catamenial pneumothorax.6 The recurrence rate of catamenial pneumothorax following surgery is between 9% and 40%.9 The current standard therapeutic approach for catamenial pneumothorax is to combine surgery with pharmacological treatment.10 Hormonal therapies, such as gonadotropin-releasing hormone agonists or oral contraceptives, may be used to suppress ovulation and prevent recurrence. Furthermore, research indicates that hysterectomy combined with bilateral salpingo-oophorectomy could serve as a definitive treatment for catamenial pneumothorax.6 In our case, the patient underwent a total hysterectomy to remove the source of endometrial tissue and subsequent thoracoscopic treatment to remove endometrial tissue from the pleural cavity. This approach successfully prevented further pneumothorax episodes.

Catamenial pneumothorax is a rare but potentially life-threatening condition that affects women of reproductive age and requires a high index of suspicion for diagnosis. The diagnosis is based on the patient's clinical history, physical examination findings, and imaging studies. The presence of endometrial tissue in the pleural cavity is the definitive diagnostic criterion. Treatment options include observation, thoracentesis or chest tube placement, surgery, and hormonal therapy. This case highlights the importance of a multidisciplinary approach to the management of catamenial pneumothorax involving pulmonologists, gynecologists, and thoracic surgeons. Further studies are needed to optimize the management of this rare condition. CONFLICT OF INTEREST The authors have no conflicts of interest to declare. Chest X-ray showing the persistence of pneumothorax in the right hemithorax after chest tube placement. Figure 2. Thoracoscopic image showing the diaphragm intraoperatively. The image shows fenestrations (arrows) in the diaphragm that were identified and subsequently repaired with 4-0 polypropylene sutures. Figure 3. Chest X-ray post-thoracoscopy, after closure of the diaphragmatic hole, and total pleurectomy. A discrete minimal apical pneumothorax is evident. Figure 4. Chest X-ray at the time of patient discharge showing complete expansion of the right lung with no residual pneumothorax. [1] LukacS, SchmidM, PfisterK, JanniW, SchäfflerH, DayanD. , 'Extragenital endometriosis in the differential diagnosis of non-gynecological diseases' () Dtsch Arztebl Int. 2022 May 20;119(20):361–67. doi: 10.3238/arztebl.m2022.0176. . ArakawaS, MatsudairaH, NodaY, YamashitaM, HiranoJ, OgawaM, , 'Catamenial pneumothorax with partial liver herniation due to diaphragmatic laceration: A case report and literature review' () J Cardiothorac Surg. 2021 Mar 17;16(1):23. doi: 10.1186/s13019-021-01407-z. . MiedziarekC, KasprzykM. , 'Catamenial pneumothorax – Are there benefits of cooperation between the surgeon and the gynaecologist? ' () Prz Menopauzalny. 2022 Mar;21(1):69–72. doi: 10.5114/pm.2022.113776. . DongB, WuC-L, ShengY-L, WuB, YeG-C, LiuY-F, et al. , 'Catamenial pneumothorax with bubbling up on the diaphragmatic defects: A case report' () BMC Womens Health. 2021 Apr 20;21(1):167. doi: 10.1186/s12905-021-01318-0. . OkyereI, GloverPSK, ForsonPK, OkyereP, Blood-DzrakuD. , 'Catamenial pneumothorax in Ghana: Case report and literature review' () Pan Afr Med J. 2019 Aug 6;33:287. doi: 10.11604/pamj.2019.33.287.14187. .

Catamenial pneumothorax is a rare but potentially life-threatening condition that affects women of reproductive age. It is characterized by the recurrent occurrence of pneumothorax during or around the menstrual period. The pathophysiology of catamenial pneumothorax is not well understood, but is thought to be related to the presence of endometrial tissue in the pleural cavity. Case Presentation: A 42-year-old woman was referred to our outpatient department for further thoracic surgical treatment. She already had two previous episodes of pneumothorax that occurred during her menstrual period. A chest X-ray revealed a right-sided pneumothorax and she was admitted to the hospital where a chest tube was placed to treat the pneumothorax. CT scan revealed the presence of endometrial tissue under the right diaphragm. The patient underwent a total laparoscopic hysterectomy to remove the source of endometrial tissue. The subdiaphragmatic endometrial tissue was also removed. She subsequently underwent thoracoscopic treatment to treat the pneumothorax.

The aim of this study was to review the clinical features, diagnostic criteria, and treatment options for catamenial pneumothorax. In addition, the study highlights the importance of a multidisciplinary approach to the management of catamenial pneumothorax, involving pulmonologists, gynecologists, and thoracic surgeons. Further studies are needed to optimize the management of this rare condition. Catamenial pneumothorax is a rare but potentially life-threatening condition that affects women of reproductive age. It is characterized by the recurrent occurrence of pneumothorax during or around the menstrual period.1 The pathophysiology of catamenial pneumothorax is not well understood, but is thought to be related to the presence of endometrial tissue in the pleural cavity.2 Here, we present the case of a 42-year-old woman with recurrent catamenial pneumothorax who underwent a hysterectomy and subsequent thoracoscopic treatment. This article also aims to review the clinical features, diagnostic criteria, and treatment options for catamenial pneumothorax. CASE PRESENTATION A 42-year-old woman was referred to our outpatient department for further thoracic surgical treatment. She had two previous episodes of pneumothorax that occurred during her menstrual period. On physical examination, she had decreased breath sounds on the right side of her chest on both occasions. A chest X-ray revealed a right-sided pneumothorax, and she was admitted to the hospital where a chest tube was placed to treat the pneumothorax (Figure 1). Further investigations, including a computed tomography (CT) scan, had already been performed at another hospital abroad to identify the underlying cause of the recurrent pneumothorax. The previously performed CT scan had revealed the presence of endometrial tissue under the right diaphragm. The patient had undergone a total laparoscopic hysterectomy two months ago to remove the source of endometrial tissue. The subdiaphragmatic endometrial tissue had also been removed. She subsequently underwent thoracoscopic treatment to treat the pneumothorax. Due to the patient's young age and limited visibility of endometrial tissue on CT scans, a preoperative CT scan was not performed, and the decision was made to proceed directly to thoracoscopy, as a preoperative CT scan would not change the therapeutic approach. Thoracoscopy did not identify endometrial tissue in the pleural cavity. Two holes in the diaphragm were identified and sutured endoscopically with 4-0 polypropylene sutures (Figure 2). A total thoracoscopic pleurectomy was also performed (Figure 3). The postoperative course was uneventful, and the patient remained stable throughout her hospital stay and was discharged home on oral contraceptives and close follow-up by her pulmonologist and gynecologist (Figure 4). Histology showed no signs of endometriosis. Follow-up examinations by the pulmonologist revealed no further episodes of pneumothorax.

Catamenial pneumothorax is a rare medical condition that accounts for approximately 3–6% of all cases of spontaneous pneumothorax.3 Women of reproductive age are primarily affected, with an average onset age of 35 years.2 Catamenial pneumothorax can be the first manifestation of the thoracic endometriosis syndrome (TES), and is attributed to the presence of endometriotic lesions in the pulmonary and pleural tissues.4 Endometriosis, a prevalent clinical disease among women, usually occurs in the pelvic region but in rare cases can also manifest in extrapelvic regions including the abdominal wall, vulva, perineum, liver, skin, central nervous system, and thoracic cavity.5 The clinical manifestations of thoracic endometriosis include catamenial pneumothorax, hemothorax, and hemoptysis. Patients with these manifestations of spontaneous pneumothorax are often diagnosed without prior comprehensive gynecological evaluation to exclude the existence of catamenial pneumothorax.6 The pathophysiology of catamenial pneumothorax remains a subject of limited understanding.7 Various theories concerning catamenial pneumothorax have been postulated.8 The main theories include the following:2 • Physiological hypothesis. • Metastatic or lymphovascular microembolization theory. • Transgenital–transdiaphragmatic passage of air theory. • Migration theory. According to the “physiological hypothesis”, prostaglandin F2 levels increased during menstruation can trigger vasoconstriction and bronchospasm, leading to alveolar rupture and consequently pneumothorax.2 According to the “metastatic or lymphovascular microembolization theory”, endometrial tissue is transported to the lungs via the venous or lymphatic system. Subsequently, periodic menstruation-induced necrosis of endometriotic lesions adjacent to the visceral pleura results in pneumothorax. In certain cases, hemoptysis can also occur when the endometriotic foci are centrally located.2 The “transgenital–transdiaphragmatic passage of air theory” suggests that atmospheric air travels from the vagina to the uterus, then through the fallopian tubes into the peritoneal cavity, and finally enters the thoracic cavity via diaphragmatic defects.9 According to the “migration theory”, endometrial cells migrate from the lower pelvic region through the peritoneal circulation, eventually reaching subdiaphragmatic areas, predominantly on the right side. This is attributed to the clockwise circulation of peritoneal fluid. Periodic necrosis of endometrial tissue leads to diaphragmatic penetration, resulting in the entry of endometrial tissue into the thoracic cavity. This endometrial tissue implants itself onto the visceral pleura and causes alveolar rupture through cyclical necrosis, leading to pneumothorax.2 Symptoms of catamenial pneumothorax typically manifest around the menstrual period (from 24 hours before to 72 hours after the onset of menstrual bleeding), but they can also occur at other times. Catamenial pneumothorax typically presents with an abrupt onset of chest pain and dyspnea.1,2 A common symptom is pain associated with the menstrual cycle, often localized on the right side of the chest, in the scapular or shoulder area. This localization can be attributed to the shared C5 spinal segment innervation between the diaphragm and these regions. Pneumothorax mostly occurs on the right side, with less frequent occurrences on the left side or bilaterally. The exact reason for this preference is not yet fully understood. Patients may also experience coughing and fatigue.1 The diagnosis of catamenial pneumothorax is based on the patient's clinical history, physical examination findings, and imaging studies. Various imaging modalities such as chest X-ray, CT scans, or magnetic resonance imaging (MRI) are used to confirm the diagnosis.4 The definitive diagnostic criterion is evidence of the presence of endometrial tissue in the pleural cavity, typically by biopsy or thoracoscopy1 While MRI appears to be more accurate in detecting endometriotic lesions in the abdomen, it is also used as a diagnostic tool to identify endometriotic lesions associated with TES. It exhibits particular precision in detecting endometriotic lesions in the diaphragmatic and pericardial regions, but is less accurate in diagnosing visceral, parietal pleural, or bronchopulmonary endometriosis.4 Treatment for catamenial pneumothorax depends on the severity of the condition. Small pneumothoraxes may resolve spontaneously, but larger ones require intervention. Acute pneumothorax can be treated by thoracentesis or chest tube placement. For recurrent or persistent pneumothorax, surgical interventions such as video-assisted thoracic surgery (VATS) or thoracotomy may be warranted. VATS is typically the preferred treatment, while thoracotomy remains an option, especially in reoperations.2 Surgery is ideally performed during the menstrual period, as symptoms and pathological findings are more conspicuous.7 Intraoperatively, endometrial implants were observed in 59.3% of patients, and histopathology confirmed the presence of thoracic endometriosis in 52.3% of patients diagnosed with catamenial pneumothorax.6 The recurrence rate of catamenial pneumothorax following surgery is between 9% and 40%.9 The current standard therapeutic approach for catamenial pneumothorax is to combine surgery with pharmacological treatment.10 Hormonal therapies, such as gonadotropin-releasing hormone agonists or oral contraceptives, may be used to suppress ovulation and prevent recurrence. Furthermore, research indicates that hysterectomy combined with bilateral salpingo-oophorectomy could serve as a definitive treatment for catamenial pneumothorax.6 In our case, the patient underwent a total hysterectomy to remove the source of endometrial tissue and subsequent thoracoscopic treatment to remove endometrial tissue from the pleural cavity. This approach successfully prevented further pneumothorax episodes.

Catamenial pneumothorax is a rare but potentially life-threatening condition that affects women of reproductive age and requires a high index of suspicion for diagnosis. The diagnosis is based on the patient's clinical history, physical examination findings, and imaging studies. The presence of endometrial tissue in the pleural cavity is the definitive diagnostic criterion. Treatment options include observation, thoracentesis or chest tube placement, surgery, and hormonal therapy. This case highlights the importance of a multidisciplinary approach to the management of catamenial pneumothorax involving pulmonologists, gynecologists, and thoracic surgeons. Further studies are needed to optimize the management of this rare condition. CONFLICT OF INTEREST The authors have no conflicts of interest to declare. Chest X-ray showing the persistence of pneumothorax in the right hemithorax after chest tube placement. Figure 2. Thoracoscopic image showing the diaphragm intraoperatively. The image shows fenestrations (arrows) in the diaphragm that were identified and subsequently repaired with 4-0 polypropylene sutures. Figure 3. Chest X-ray post-thoracoscopy, after closure of the diaphragmatic hole, and total pleurectomy. A discrete minimal apical pneumothorax is evident. Figure 4. Chest X-ray at the time of patient discharge showing complete expansion of the right lung with no residual pneumothorax. [1] LukacS, SchmidM, PfisterK, JanniW, SchäfflerH, DayanD. , 'Extragenital endometriosis in the differential diagnosis of non-gynecological diseases' () Dtsch Arztebl Int. 2022 May 20;119(20):361–67. doi: 10.3238/arztebl.m2022.0176. . ArakawaS, MatsudairaH, NodaY, YamashitaM, HiranoJ, OgawaM, , 'Catamenial pneumothorax with partial liver herniation due to diaphragmatic laceration: A case report and literature review' () J Cardiothorac Surg. 2021 Mar 17;16(1):23. doi: 10.1186/s13019-021-01407-z. . MiedziarekC, KasprzykM. , 'Catamenial pneumothorax – Are there benefits of cooperation between the surgeon and the gynaecologist? ' () Prz Menopauzalny. 2022 Mar;21(1):69–72. doi: 10.5114/pm.2022.113776. . DongB, WuC-L, ShengY-L, WuB, YeG-C, LiuY-F, et al. , 'Catamenial pneumothorax with bubbling up on the diaphragmatic defects: A case report' () BMC Womens Health. 2021 Apr 20;21(1):167. doi: 10.1186/s12905-021-01318-0. . OkyereI, GloverPSK, ForsonPK, OkyereP, Blood-DzrakuD. , 'Catamenial pneumothorax in Ghana: Case report and literature review' () Pan Afr Med J. 2019 Aug 6;33:287. doi: 10.11604/pamj.2019.33.287.14187. .