Catamenial Haemorrhagic Pleural Effusion Caused by Thoracic Endometriosis, which was Controlled by Surgery Undertaken after Failed Medical Management.

Haemorrhagic pleural effusion can be a challenging diagnosis that requires a thorough investigation and sometimes a multidisciplinary team of physicians to reach the underlying aetiology. Causes can include pulmonary malignancy, pulmonary infections, connective tissue diseases, asbestos associated, intra-abdominal conditions such as pancreatitis and ovarian tumours, cardiovascular disorders such as ruptured aneurysms and pulmonary infarction, as well as other miscellaneous causes. One such cause is endometriosis in the thoracic cavity. Endometriosis is a chronic illness associated with the occurrence of endometrial tissue outside the endometrium. Insertion of endometrial tissue in the thoracic cavity is rare, with only a few cases described. This case report gives detail of a 30-year-old nulligravida suspected of having thoracic endometriosis following a history of catamenial dyspnoea and associated pleural effusion. The diagnosis was confirmed through the histopathological study of tissue obtained via thoracoscopic surgery. Excision of the endometrial tissue was done, and the patient then continued medical treatment with progestins and gonadotrophin-releasing hormone (GnRH) agonists. Following therapy, the index patient was asymptomatic. A multidisciplinary approach is often needed in the diagnosis and management of thoracic endometriosis, involving both medical and surgical specialities. Minimally invasive surgery is the gold standard of diagnosis, allowing for direct visualisation of implants and nodules and should be followed by medical treatment to reduce the risk of recurrence. Medical therapy alone is associated with higher rates of recurrence. Physicians must have a high degree of suspicion as thoracic endometriosis is a disease that can often be missed. LEARNING POINTS: Thoracic endometriosis syndrome is a rare but significant cause of haemorrhagic pleural effusion in women of childbearing age.Diagnosis and treatment can be challenging, and a multidisciplinary approach has been found to improve outcomes.

A surgical series on endometriosis-related diaphragmatic hernia.

BACKGROUND: Thoracic endometriosis syndrome gives rise to various clinical and radiological manifestations. We reviewed the records of patients operated for intrathoracic migration of abdominal viscera through a diaphragmatic hernia secondary to thoracic endometriosis. METHODS: We retrospectively reviewed the single-center prospective collected database of all patients operated for thoracic endometriosis during the twenty years. All cases in which an abdominal organ was found to be herniated into the thoracic cavity were retrieved. Clinical and pathological data are presented and analyzed. RESULTS: Twenty women of median age 36 (range 25-58) years were operated for endometriosis-related diaphragmatic hernia. The hernia was diagnosed concomitantly with endometriosis-related pneumothorax in 13 cases and during the exploration of catamenial thoracic pain in seven cases. There were 18 cases on the right side and two cases on the left side. The median diameter of the hernia was 8 cm (2.5-20 cm). In seventeen cases, the hernia was repaired by direct suture, and in three cases a heterologous prosthesis was positioned. At follow-up, two patients had an episode of recurrent pneumothorax. CONCLUSIONS: Diaphragmatic hernia should be ruled out in the presence of endometriosis-related pneumothorax or catamenial thoracic pain. Surgery is indicated to make a pathological diagnosis, restore anatomy, and prevent recurrence in patients presenting with pneumothorax.

Spontaneous recurrent menstrual pneumothorax: a case report.

Introduction and importance: Endometriosis is most commonly found in the pelvic area, ~12% of people have it in other areas or organs, which is known as extrapelvic endometriosis. Thoracic endometriosis, which is also classified as extrapelvic endometriosis, manifests with four distinct forms: catamenial pneumothorax, catamenial hemothorax, catamenial hemoptysis, or lung nodules. Catamenial pneumothorax is the most common clinical symptom of these; however, it is frequently neglected by clinicians and goes undiagnosed and untreated. As a result, it is critical to raise awareness of this medical condition among clinicians. Case presentation: The authors present a case report of a 34-year-old woman of reproductive age who had recurrent episodes of spontaneous pneumothorax during menstruation and underwent treatment with thoracoscopic surgery as well as gynaecological hormonal drugs including oral progesterone and dienogest throughout this time. Based on her symptoms, a catamenial pneumothorax caused by thoracic endometriosis was suspected. Clinical discussion: The clinical symptoms, pathogenesis, diagnosis, and treatment of Catamenial Pneumothorax are analyzed. Furthermore, the usage of gynaecological hormone medications in this condition has been discussed. The mechanisms of oral contraceptives and progestin-based medications are evaluated by comparing the patient's treatment process, highlighting their pros and cons. Conclusions: Thoracoscopic surgery combined with postoperative gynaecological hormonal medications may be the most effective treatment for this issue. Several gynaecological hormonal medicines are available, each of which has its own set of pros and cons, and must be thoroughly evaluated as well as correctly tailored to the patient's specific circumstances to have a positive therapeutic outcome.

Endometriosis first presenting in pleural fluid cytology.

Most patients with thoracic endometriosis present with catamenial pneumothorax, a rare condition in which recurrent episodes occur within 72 h before or after the start of menstruation. We report a case of thoracic endometriosis presenting with recurrent bloody pleural effusions without pneumothorax diagnosed on pleural fluid cytology. We describe the cytomorphology and immunoprofile of thoracic endometriosis and discuss the differential diagnoses, including neoplastic processes. We also highlight the importance of communication with clinicians for timeliness of diagnosis and treatment, especially when thoracic endometriosis is not suspected.

Diaphragmatic Endometriosis Presenting as Recurrent Catamenial Pneumothorax: A Case Report.

Catamenial pneumothorax is one of the most common extra-pelvic presentations of endometriosis, with the gastrointestinal tract being the most common location. Catamenial pneumothorax is defined as spontaneous recurrent pneumothorax occurring in women of reproductive age in a temporal relationship with menses. Symptoms include dyspnea, sharp chest pain, and hypoxemia. A much rarer presentation is the involvement of endometriosis with the diaphragm. In this case, we present a 31-year-old female who presented with signs of pneumothorax. She has had multiple episodes leading to suspicion of catamenial pneumothorax. However, it wasn't until her surgery that the extent of diaphragmatic involvement, characterized by numerous holes secondary to endometriosis, was discovered. She was surgically treated, which led to a drastic improvement in symptoms and a reduction in subsequent episodes. We hope that this case can add to the current limited literature on diaphragmatic endometriosis cases. Since this patient presented with mainly catamenial pneumothorax symptoms, we urge clinicians to still consider diaphragmatic involvement as a primary cause in patients with recurrent episodes of pneumothorax.

Postoperative hormonal therapies reduce the recurrence of thoracic endometriosis-related pneumothorax.

OBJECTIVES: Thoracic endometriosis-related pneumothorax (TERP) frequently recurs even after surgery. Meanwhile, postoperative hormonal therapies (HTx) are believed to be effective for pelvic endometriosis. Therefore, we evaluated the relationship between postoperative TERP recurrence and postoperative HTx in a retrospective observational study. METHODS: We retrospectively reviewed the data of patients with TERP who underwent the first video-assisted thoracoscopic surgery between January 2011 and February 2022. RESULTS: Of the 248 patients eligible for this study, 67 (27.0%) experienced postoperative TERP recurrence. Postoperative HTx were administered to 70 patients (28.2%). Dienogest was the most frequently administered drug, given to 56.7% of patients. Following univariable analysis, postoperative hormonal therapies was closely related to reduce postoperative recurrence (P = 0.003). Likewise, the multivariable analysis revealed postoperative hormonal therapies were significantly associated with the risk reduction of recurrence (hazard ratio 0.28, P < 0.001). CONCLUSIONS: Postoperative HTx reduced TERP recurrence. We hypothesize that HTx may control residual endometrial tissues to avoid TERP if pleural endometrial tissues are resected as much as possible.

Thoracic Endometriosis Syndrome (TES) in Martinique, a French West Indies Island.

INTRODUCTION: Endometriosis is a female disease that affects 5-10% of women of childbearing age, with predominantly pelvic manifestations. It is currently declared as a public health priority in France. Thoracic endometriosis syndrome (TES) is the most common extra-pelvic manifestation. OBJECTIVE: The objective of this study was to describe the epidemiological and clinical characteristics, and outcomes of patients with TES in Martinique. PATIENTS AND METHODS: We performed a descriptive, retrospective study including all patients managed at the University Hospital of Martinique for TES between 1 January 2004 and 31 December 2020. RESULTS: During the study period, we identified 479 cases of pneumothorax, of which 212 were women (44%). Sixty-three patients (30% of all female pneumothorax) were catamenial pneumothorax (CP) including 49 pneumothoraxes alone (78% of catamenial pneumothorax) and 14 hemopneumothorax (22% of catamenial pneumothorax). There were 71 cases of TES, including 49 pneumothoraxes (69%), 14 hemopneumothoraxes (20%) and 8 hemothorax (11%). The annual incidence of TES was 1.1 cases/100,000 inhabitants. The prevalence of TES was 1.2/1000 women aged from 15 to 45 years and the annual incidence of TES for this group was 6.9/100,000. The annual incidence of CP was 1 case/100,000 inhabitants. The average age at diagnosis was 36 ± 6 years. Eight patients (11%) had no prior diagnosis of pelvic endometriosis (PE). The mean age at pelvic endometriosis diagnosis was 29 ± 6 years. The mean time from symptom onset to diagnosis was 24 ± 50 weeks, and 53 ± 123 days from diagnosis to surgery. Thirty-two patients (47%) had prior abdominopelvic surgery. Seventeen patients (24%) presented other extra-pelvic localizations. When it came to management, 69/71 patients (97%) underwent surgery. Diaphragmatic nodules or perforations were found in 68/69 patients (98.5%). Histological confirmation was obtained in 55/65 patients who underwent resection (84.6%). Forty-four patients (62%) experienced recurrence. The mean time from the initial treatment to recurrence was 20 ± 33 months. The recurrence rate was 16/19 (84.2%) in patients who received medical therapy only, 11/17 (64.7%) in patients treated by surgery alone, and 17/31 (51.8%) in patients treated with surgery and medical therapy (p = 0.03). CONCLUSIONS: We observed a very high incidence of TES in Martinique. The factors associated with this high incidence in this specific geographical area remain to be elucidated. The frequency of recurrence was lower in patients who received both hormone therapy and surgery.

Catamenial Pneumothorax in a Patient with Endometriosis: A Case Report.

Pelvic pain is a common presentation to the emergency department (ED). For female patients, endometriosis can be difficult to diagnose and can have life-threatening complications if missed. In this case report, we present a case of a patient initially presenting to the ED with a few days of crampy lower abdominal pain. After initial imaging, she was found to have a large pelvic hematoma with concern for active extravasation and a large hemothorax. After further evaluation, she was suspected of having endometriosis leading to thoracic endometriosis and a catamenial pneumothorax. Although endometriosis is not typically an emergent diagnosis, the complications of significant endometrial tissue spread can cause life-threatening impacts. Clinicians should consider complications of endometriosis in females of menstruating age.

A Rare Case of Catamenial Pneumothorax and a Review of the Current Literature.

A 34-year-old female smoker, with a history of pelvic endometriosis, presented with initial symptoms of shortness of breath and a choking sensation. She was found to have a right pneumothorax on chest x-ray. Over the next eight months, she ultimately underwent three tube thoracostomies, two video-assisted thoracoscopic surgeries (VATS), wedge resection, and repeated pleurodesis due to pneumothorax recurrence. She was seen multiple times post-surgically with the focus of treatment being smoking cessation rather than contraceptive therapy, despite an early follow-up visit noting that the initial symptoms coincided with her menstruation. The purpose of this article is to bring attention to this rarely diagnosed condition. With added awareness and understanding of the underlying causes and available treatments, medical providers could likely spare many women from similar experiences and dramatically improve the quality of their lives.

Pleural effusion secondary to endometriosis: A systematic review.

BACKGROUND: Endometriosis-associated pleural effusion is a rare occurrence with poorly defined clinical characteristics. METHODS: A systematic review was performed to examine all articles on endometriosis-associated pleural effusion extracted from 4 databases (PubMed, Embase, Web of Science and Scopus) from inception until November 2022. RESULTS: A total of 142 articles (isolated cases and small retrospective series) involving 176 patients (median age 33 years) with endometriosis-associated pleural effusion were included. The most frequent symptoms were dyspnea (67%), chest pain (55%) and abdominal pain (40%). Pleural effusion was predominantly unilateral (89%), right-sided (88.5%) and massive (56%). Ascites was evident in 42% of the cases. Pleural fluid had a bloody appearance in 99% of cases and always met the exudate criteria. Pleural fluid cytology identified only 9% of the patients, with pleural biopsy being the most common diagnostic procedure (74%). Most patients were treated with hormones (76%), thoracic surgery (60%) and abdominal surgery (27%). Effusion recurrence was observed in 26% of cases after a median follow-up of 1 year. CONCLUSIONS: The presence of right-sided hemorrhagic pleural effusion in a young woman warrants an assessment for the possibility of endometriosis. Despite conventional treatment, effusion recurs in approximately a quarter of patients.

Catamenial pneumothorax: Not only VATS diagnosis.

Background: Catamenial pneumothorax (CP) is a rare type of spontaneous, recurring pneumothorax occurring in women, from the day before menstruation until 72 hours after its beginning. Conservative treatment is generally associated with recurrence of CP. Video-assisted thoracic surgery (VATS) approach allows not only to obtain diagnosis but also to guide definitive treatment of causing lesions, such as ectopic endometrial implants or diaphragmatic defects and fenestrations. We report our experience in VATS management of CP to focus on its role in CP. Materials and methods: In this retrospective observational study, we collected data from women referred to our center for CP, from January 2019 to April 2022. All patients underwent VATS approach, with muscle-sparing thoracotomy when diaphragmatic fenestrations were detected, to perform selective diaphragmatic plication and/or partial diaphragmatic resection. Results were analyzed in terms of pneumothorax recurrence after surgical treatment. All patients were referred to gynecologists for medical therapy. Results: Eight women (median age 36 years, range: 21-45), all with right side CP, were included; three already had pelvic endometriosis and two had already undergone lung apicectomy at other institutions. VATS allowed us to detect diaphragmatic fenestrations in seven patients (87.5%) and apical bullae in five (62.5%). Apicectomy was performed in five cases (62.5%), selective diaphragmatic plication in two (25%), and partial diaphragmatic resection in five (62.5%). Chemical pleurodesis with talc was performed in all to minimize the risk of recurrence. Pathological diagnosis of endometriosis on the resected diaphragm was achieved in five patients (62.5%). No recurrence occurred, except for one woman who stopped medical treatment for endometriosis. Conclusions: In the management of patients with CP, VATS should be recommended not only to obtain an explorative diagnosis of ectopic endometrial implants or diaphragmatic fenestrations but also to allow the most appropriate surgical treatment and obtain pathological specimens for confirmation and definitive diagnosis of thoracic endometriosis. Medical therapy to achieve ovarian rest is mandatory in the postoperative period and should not be discontinued.

Pleural Endometriosis: An Atypical Cause of Hemoptysis.

Endometriosis is a benign, inflammatory disease characterized by the presence of dysfunctional endometrial tissue outside the uterus. Typically confined to the pelvis, endometriosis is frequently associated with pain, dysmenorrhea, and infertility. Rarely, endometrial tissue has been documented to implant within the lung parenchyma and involve both parietal and visceral pleura of the thorax. Manifestations of thoracic endometriosis include catamenial pneumothorax, hemothorax, and hemoptysis. We present a case of thoracic endometriosis in a 43-year-old female who was found to have a loculated pleural effusion with an associated pleural nodule after gynecologic surgery. The patient underwent thoracotomy, decortication, and nodule excision. Pathology of the pleural nodule showed evidence of endometrial tissue within the parietal pleural. Thoracic endometriosis is a medical problem that is frequently undiagnosed and encountered by the practicing surgeon. Early diagnosis reduces both disease progression and late complications, allowing for early initiation of appropriate medical and surgical therapy.

Hemotórax catamenial por implantes endometriales diafragmáticos y pulmonares / Catamenial hemothorax due to diaphragmatic and pulmonary endometrial implants

La endometriosis torácica afecta principalmente a mujeres jóvenes y es originada por la migración de tejido endometrial a la cavidad torácica. Se suele presentar como neumotórax, hemoptisis, hemotórax o nódulos pulmonares. El tratamiento hormonal es el enfoque de primera línea. La cirugía actúa como herramienta diagnóstico-terapéutica y sin embargo el diagnóstico definitivo es histológico. Presentamos el caso de una mujer con diagnóstico de endometriosis pélvica en tratamiento médico a quien se le detecta hemotórax, siendo tratada con cirugía que permitió visualizar implantes pulmonares y diafragmáticos. (AU)

Thoracic endometriosis mainly affects young women and is caused by the migration of endometrial tissue into the thoracic cavity. It usually presents as pneumothorax, hemoptysis, hemothorax, or pulmonary nodules. Hormone treatment is the first line approach. Surgery acts as a diagnostic-therapeutic tool and yet the definitive diagnosis is histological. We present the case of a woman with a diagnosis of pelvic endometriosis undergoing medical treatment who was detected with a hemothorax, being treated with surgery that allowed the visualization of pulmonary and diaphragmatic implants. (AU)

Hemoneumotórax espontaneo, una manifestación inusual de la endometriosis pleural / Spontaneous hemopneumothorax, an unusual manifestation of pleural endometriosis

El hemotórax y neumotórax es una condición usualmente relacionada con etiología traumática, infecciosa, inflamatoria o coagulopatía. La aparición espontanea en pacientes jóvenes precisa la búsqueda de etiologías diferenciales. La expresión clínica de la endometriosis pleural incluye neumotórax, hemotórax, hemoptisis y nódulos pulmonares. La radiografía de tórax, la tomografía computarizada, la resonancia magnética nuclear, la broncoscopia y la biopsia se constituyen como el pilar del diagnóstico. Presentamos un caso de un paciente joven con disnea y hemo-neumotórax espontaneo con diagnóstico de endometriosis pleural. (AU)

Hemothorax and pneumothorax is a condition usually related to traumatic, infectious, inflammatory or coagulopathy etiology. The spontaneous appearance in young patients requires the search for differential etiologies. The clinical expression of pleural endometriosis includes pneumothorax, hemothorax, hemoptysis, and pulmonary nodules. Chest X-ray, computed tomography, nuclear magnetic resonance, bronchoscopy and biopsy constitute the mainstay of diagnosis. We present a case of a young patient with dyspnea and spontaneous hemo-pneumothorax diagnosed with pleural endometriosis. (AU)

Thoracic endometriosis masquerading as Meigs' syndrome in a young woman: A case report and literature review.

Thoracic endometriosis is an exceedingly rare condition characterized by the presence of endometriotic deposits on the diaphragm, lungs or pleural space. Patients may present with massive hemothorax, pneumothorax, hemoptysis or pulmonary nodules. It is a complex condition that often proves to be a diagnostic challenge, resulting in under-diagnosis, delays in treatment and significant morbidity in women of reproductive age. We report a case of endometriosis causing massive pleural effusion and ascites, with a left adnexal fibroid mass mimicking Meigs' syndrome in a nulliparous woman in her late 30s. The patient was successfully managed with hormonal therapy following fertility-sparing surgical treatment. This case highlights the diagnostic and therapeutic challenges associated with thoracic endometriosis because of its close resemblance to more sinister gynecological conditions. Hormonal therapy is the long-term treatment of choice in patients with thoracic endometriosis to reduce the risk of symptom recurrence and preserve fertility.

Catamenial pneumothorax: a rare manifestation of endometriosis.

Endometriosis is a common gynecological disease that primarily affects premenopausal women. It is mainly found in the pelvis but may be found at several extrapelvic locations. Thoracic endometriosis is a rare extrapelvic location of endometriosis and the leading cause of catamenial pneumothorax. We describe the case of a 35-year-old woman with a background of pelvic pain presenting to the emergency department with chest pain and dyspnea. The chest X-ray in the emergency department showed a large right-sided pneumothorax. Further imaging studies during patient evaluation revealed extensive fibrotic changes in the pelvis and well-defined solid nodules with high signal on T2 and T1-weighted images on MRI in abdominal and thoracic locations, rendering the diagnosis of a catamenial pneumothorax in a patient with pelvic, abdominal and thoracic endometriosis.

Shortness of breath in a young lady, rare case report of thoracic endometriosis.

INTRODUCTION AND IMPORTANCE: Endometrial glandular tissue can implant in the thorax of women suffering from endometriosis. The clinical presentation is depends on site of implantation. Complications include pneumothorax, pneumohemothorax or hemothorax. CASE PRESENTATION: A 31 year old woman with history of infertility presented with shortness of breath and was found to have a significant right sided pneumohemothorax. Drainage was done followed by chemical pleurodesis using bleomycin with resolution of symptoms on her follow up. CLINICAL DISCUSSION: Thoracic endometriosis tend to present with chronic or sub-acute symptoms which are non-specific symptoms leading to late diagnosis. Video Assisted Thoracoscopic surgery offer both diagnostic and therapeutic in thoracic endometriosis. However in limited settings chemical pleurodesis can be carried out done to prevent recurrence of shortness of breath due to thoracic endometriosis. CONCLUSION: Therefore, clinical suspicion of thoracic endometriosis in evaluation of shortness of breath in a young lady with history of infertility or pelvic surgery is indispensable.

Catamenial Pneumothorax in the Setting of a Recent Stroke.

Catamenial pneumothorax is a unique condition associated with thoracic endometriosis. It often presents in females of reproductive age as a recurrent pneumothorax aligned with the menstrual cycle. We present a case of a young female diagnosed with catamenial pneumothorax within one year of experiencing a stroke. The clinical presentation related to the stroke allowed for a unique diagnostic process and management considerations. The patient was successfully treated with progesterone-based contraception in the setting of an estrogen contraindication.

[Mystery of endometriosis - catamenial pneumothorax]. / Zagadki endometrioza ­ katamenial'nyi pnevmotoraks.

The review is devoted to clinical picture, main theories of pathogenesis, traditional and innovative methods of diagnosis and surgical treatment of catamenial pneumothorax. Currently, clinicians prefer magnetic resonance imaging and thoracoscopy for diagnosis of this disease. Various researchers are actively searching for laboratory diagnostic methods that can confirm endometrioid nature of pneumothorax. Treatment and prevention of catamenial pneumothorax are a controversial issue depending both clinical picture and preferences of the attending physician. Currently, the majority of authors believe that hormonal therapy combined with diaphragm resection ensures optimal anti-relapse effect in patients with thoracic endometriosis.

Catamenial Pneumothorax as the First Expression of Thoracic Endometriosis Syndrome and Pelvic Endometriosis.

OBJECTIVE: The menstrual-related catamenial pneumothorax (CP) can be the first expression of thoracic endometriosis syndrome (TES), which is the presence of endometriotic lesions in the lungs and pleura, and pelvic endometriosis (PE). This study aims to analyze our experience with this specific correlation describing our multidisciplinary approach to CP. METHODS: Hospital records of 32 women, operated for CP at our Department from January 2001 to December 2021 were reviewed. Surgical treatment consisted of videothoracoscopy and laparoscopy when indicated. RESULTS: TES and PE were diagnosed in 13 (40.6%) and 12 (37.5%) women, respectively. The association of TES and PE was present in 11 cases (34%). Fifteen patients (46.9%) underwent laparoscopy, of which 11 concurrently with videothoracoscopy. Most of the patients affected had stage III-IV endometriosis (40.6%). All patients received hormonal therapy after surgery. Five patients with PE conceived spontaneously resulting in six live births. The mean follow-up was 117 ± 71 months (range 8-244). Pneumothorax recurrence occurred in six patients (18.8%). At present, all women are asymptomatic, with no sign of pneumothorax recurrence. CONCLUSIONS: CP might be the first expression of TES and/or PE. A multidisciplinary approach is advocated for optimal management of the disease.