Video-assisted surgical diagnosis and pleural adhesion management in catamenial pneumothorax: A case and literature review

A 32‐year‐old woman came to the emergency ward with a chief complaint of shortness of breath and right‐sided chest pain for 6 h before admission, which worsened with physical activity. The patient also experienced localized lower abdominal pain. She claimed there were no cough, fever, weight loss, and a prior history of chest trauma, tuberculosis, and smoking. She was nulliparous, had a medical history of pelvic endometriosis 2 years ago, and had already undergone a left salpingectomy. The patient was also on the second day of her menstrual cycle when the symptoms occurred. A previous chest x‐ray (CXR) showed right pneumothorax without a mediastinal shift (Figure  1A ). The radiological findings showed: a large right pneumothorax at the emergency ward admission (chest X‐ray, A); a right lung expansion along with an atelectatic component of the lower lobe on the second day of hospital stay following a chest tube insertion (chest CT‐scan, B); an adequate right lung expansion after surgery (chest X‐ray, C); and a complete right lung expansion a month after surgical and pleurodesis procedure (chest X‐ray, D) The vital signs showed a blood pressure of 133/88 mmHg, heart rate of 112 beats/minute, respiratory rate of 22 times/min, and SpO2 of 97% with a 3 lpm oxygen nasal cannula. Physical chest examination showed the right hemithorax to be left behind during inspiration, palpation showed the lower fremitus on the right side, and percussion showed a hypersonor on the right hemithorax. Clear breath sounds were absent on the right side. We inserted a right‐sided chest tube, and the chest CT scan showed expansion of the right lung with the atelectasis components (Figure  1B ). The patient underwent a video‐assisted thoracoscopy (VATS) on the seventh day of hospitalization (Figure  2A, C ). During VATS, we found multiple perforations in the tendinous part of the diaphragm. The lesions found in this patient were multiple diaphragmatic perforations (Figure  2B ) and visceral pleural blueberry‐like spots or elliptical red spots (Figure  2D ), which were characteristics of CPX. We performed a partial resection of the tendinous diaphragm and obtained a biopsy specimen from the diaphragmatic lesions. Mechanical and chemical pleurodesis was finally performed on the patient using talc pleurodesis. Post‐procedural CXR showed no pneumothorax and an intact chest tube on the right hemithorax (Figure  1C ). Two days after the surgery, the patient was discharged and received synthetic progesterone for 1 month, during which the patient did not show any sign and symptom of recurrences with CXR examination as followed (Figure  1D ). The patient did not continue the hormonal therapy at following months and until recent follow up, there is no sign of recurrency. The biopsy result (Figure  3 ) demonstrated fibro‐adipose tissue with acute perivascular inflammation with the absence of endometrial glands or stroma. The video‐assisted thoracoscopic surgery displayed: a normal right lung at its thoracic (A) and diaphragmatic (C) sides; multiple perforations in the tendinous part of the right diaphragm (white circle, B); and blueberry‐like spots in the right visceral pleura (white arrow, D) The histopathological findings obtained from the tendinous part of the right diaphragm specimens revealed connective tissue with attached nerves and congestive blood vessels. No endometrial glands or stroma were found. The conclusion was fibro‐adipose tissue with acute perivascular inflammation (Neutral buffered 10% formaldehyde fixation, H&E staining, 100× magnification)

Irandi Putra Pratomo as the principal investigator, data collector, concept writer, and manuscript reviewer; Muhammad Arza Putra as the co‐investigator, data collector, and manuscript reviewer; Lidia Giritri Bangun and Isti Mardiana Soetartio as the co‐investigators, data collectors, data analysts, and manuscript writers; and Maria Angela Putri Maharani and Irene Sinta Febriana as the co‐investigators, data analysts, and manuscript reviewers. Dicky Soehardiman and Prasenohadi Prasenohadi as the data analysts and manuscript reviewers. Tutug Kinasih as the manuscript writer. All authors have read and approved the final version.

The authors declare that appropriate written informed consent was obtained for the publication of this manuscript and accompanying images.

Catamenial pneumothorax is a rare primary spontaneous pneumothorax that mostly occurs in a productive‐age female. Catamenial pneumothorax occurs within 72 h before and after menstrual onset. The catamenial pneumothorax occurs 90% of the time on the right side. 5 Most common manifestation of TES is catamenial pneumothorax which is present in an average of 72% of cases. 3 Our patient had a first spontaneous pneumothorax on the second day of her menstrual cycle. She was also in her productive age (32 years old) and had a history of left salpingectomy due to endometriosis 3 years ago. For females of reproductive age like our patient, catamenial pneumothorax is a related diagnosis due to her condition. Some theories explained the development of thoracic foci of endometriosis and the pathogenesis between pneumothorax and menstruation. The first theory is a physiological theory that explains a high concentration of prostaglandin F2 during the menstrual cycle that may cause blood vessels and bronchioles to contract, causing an alveolar rupture and, subsequently, pneumothorax. The second theory is on the migration of endometrial cells from the uterus through the fallopian tubes into the diaphragm area. The next theory is a metastatic spread or pulmonary microembolization of endometrial cells by blood and lymph vessel. The last pathogenesis is the diaphragmatic theory of air passage through the uterus and fallopian tubes into the peritoneal cavity, then penetrating the diaphragmatic and pleural cavities. Right catamenial pneumothorax could happen when endometrial tissue circulates with the clockwise position of peritoneal fluid in the abdominal cavity down into the left peritoneal gutter up to the right gutter to the peritoneal surface of the right diaphragm. 1 , 7 Our patient had a video‐assisted thoracoscopic surgery to diagnose and treat thoracic endometriosis. From the VATS, we found blueberry spots near the diaphragm's tendons and multiple diaphragmatic perforations. Thoracic endometriosis should be diagnosed based on history taking, radiology, and direct visualization through VATS to see endometrial lesions. There is still no consensus about the fixed treatment of this problem because of the rarity, and long‐term follow‐up belongs to recurrence, but the diagnostic and therapeutic VATS remain to become the gold standard for TES, especially in CPX. 1 , 3 Based on some research, pleurodesis, especially mechanical pleurodesis, became the first choice for young patients over talc pleurodesis. Unfortunately, pleurodesis alone still has a high number of recurrences. Thoracoscopy is still the best diagnostic and treatment procedure for catamenial pneumothorax. We can evaluate directly with better visualization of the whole thoracic cavity. The VATS procedure also can perform limited resection of the diaphragm and mechanical or chemical pleurodesis. 1 , 8 Our patient got the partial resection of the pars tendinous diaphragm with mechanical and chemical pleurodesis. The surgical procedure, such as catamenial pneumothorax treatment, offers a better option to prevent the recurrence of pneumothorax. It gives a better result when a surgical procedure is performed with pleurodesis. Korom et al. collected 140 cases of postsurgical catamenial pneumothorax. Their commonest findings were diaphragmatic perforation with or without the presence of endometrial tissue. 1 , 8 Our patient did a surgical with mechanical and talc pleurodesis again while she was not menstruation since 2 days before the operation. We still found the blueberry spots near the diaphragm's tendons and multiple diaphragmatic perforations. The anatomical pathology examination result of our patient's lesion biopsy did not find the endometrial glands and stroma because the biopsy was not done during her menstrual cycle. This phenomenon is explained in the pathogenesis, which is the theory that describes prostaglandin‐induced bronchiolar constriction that causes ruptures of the alveolar in the menstrual cycle. The absence of histopathologic findings does not rule out the possibility of TES because the diagnosis of thorax endometriosis is not always by biopsy and histopathologic verification. 3 , 4 The multiple diaphragmatic perforations were the key to pathological findings. Catamenial pneumothorax may be caused by endometriotic cells actively entering the thoracic cavity leading to subpleural perforations in synchrony with the menstrual cycle. 1 Hormonal therapy controls endometrial tissues and decreases recurrence but cannot make a complete regression. The only hormonal treatment is an inadequate therapy to control catamenial pneumothorax. The best treatment to prevent a recurrence, based on many studies, is a surgical treatment with pleurodesis, besides hormonal therapy. Selective oral progesterone agonists give the same effectiveness as GnRH agonists in preventing the recurrence of endometriosis. About 32%–55% is the recurrence rate in patients with CPX related to TES, although surgery and hormonal therapy have been given. Our patient is on treatment with selective oral progesterone agonists after surgical and pleurodesis procedures. We will perform a long‐term follow‐up on our patient as an evaluation to prevent the recurrence. 3 , 7 , 9 In conclusion, catamenial pneumothorax is a rare primary spontaneous pneumothorax that mostly happens in productive‐age females within 72 h before and after the menstrual onset. Chest tube drainage is the first procedure to release the air and lead to lung expansion. Through video‐assisted thoracoscopic surgery, thoracic endometriosis should be diagnosed based on history taking, radiology, and direct visualization of endometrial lesions. VATS has become the gold standard for diagnosing and treating thoracic endometriosis, especially catamenial pneumothorax. Surgery and pleurodesis combined with hormonal therapy are the best choices for this case.

Catamenial pneumothorax (CPX) is a primary spontaneous pneumothorax commonly associated with menstrual periods. It usually happens within 72 h before or after the onset of menstruation. Maurer introduced spontaneous pneumothorax caused by endometriosis during menstruation in 1958, during which the patient had undergone the first successful surgical treatment by resection in the diaphragm defect. 1 , 2 Joseph et al. reported that thoracic endometriosis syndrome (TES) consisted of four clinical manifestations: CPX, catamenial hemothorax, endometrial lung nodules, and catamenial hemoptysis. The most common manifestation of TES is pneumothorax. 3 , 4 CPX occurs in women of reproductive age, most common in 30–40 years old, during their ovulation phase. 5 The initial treatment for the patient diagnosed with a collapsed lung is chest tube insertion. Video‐assisted thoracoscopy (VATS) is the gold standard procedure for CPX as the diagnostic and treatment to find the hallmark sign and perforation in the diaphragm. 6

None declared.