Thoracic Endometriosis Syndrome Research Articles

Establishment and Characterization of a Stromal Cell Line Derived From a Patient With Thoracic Endometriosis

Thoracic endometriosis (TE) syndrome is a clinical condition known as an extrapelvic form of endometriosis with the presence of functioning endometrial tissue involving lung parenchyma, pleura, chest wall, or diaphragm. In an effort to obtain an endometriosis ex vivo model, we established the spontaneously growing TH-EM1 cell line from endometriotic implants in lung parenchyma from a woman with TE. Maintained in long-term culture, the cells grew as large mesenchymal-like cells with a doubling time between 5 and 6 days. Treatment with medroxyprogesterone acetate (10–7 mol/L) inhibited the TH-EM1 cells growth and induced morphological changes to an epithelial-like cells. Strong expression of the nuclear estrogen receptors, progesterone receptors, and erytropoietin receptors were found in both the pulmonary implant and the TH-EM1 cells by immunohistochemical analysis. Consistent immunoreactivity of TH-EM1 cells for CD9, CD13, CD73, CD90, CD105, and CD157 was revealed by flow cytometry. Likewise, the embryonic markers, SRY-box 2 (SOX-2) and the Nanog molecules, were detected in 76% and 52% of the cells, while fetal hemoglobin and a-globin were detected in 76% and 65% of TH-EM1 cells, respectively. By RHG banding, normal metaphases were observed, while the microarray chromosomal analysis showed gains of DNA sequences located on the segments 8p23.1, 11p15.5, and 12p11.23. The described in vitro cellular model can serve as a useful tool to study the pathogenesis of endometriosis and to improve the knowledge of molecular mechanisms controlling the endometriotic cell dissemination potential.

Right hemothorax secondary to endometriosis

Endometriosis is said to have a low incidence in women with African descent. However, recent studies have shown otherwise. Thoracic involvement is rare; thoracic endometriosis syndrome mostly affects nulliparous women within the ages of 25–35 years. The diagnosis is usually missed or delayed by clinicians. We report the case of a 26-year-old female with catamenial hemothorax secondary to thoracic endometriosis. A high index of suspicion for this condition should be maintained in women of the reproductive age group presenting with dyspnea/chest pain, severe dysmenorrhea, and background history of chronic pelvic pain. A multidisciplinary approach involving the radiographers, gynecologists, pulmonologists, and the thoracic surgeons coupled with hormonal therapy and surgical treatment will go a long way in improving this disease condition and preventing reoccurrences.

Uniportal-VATS approach to thoracic endometriosis syndrome: a case report

Nowadays, endometriosis is considered a common benign disease. It affects between 6% and 10% of women of childbearing and it’s characterized by the presence of islands of endometrial-like cells outside the uterine cavity, more often located on the intraperitoneal surface of reproductive organs. The thoracic cavity is the second major district where it’s possible to find islands of endometriosis affecting the parietal and/or visceral pleural surface, the diaphragm or the lung parenchyma, therefore causing a variety of symptoms and manifestations, all together named as thoracic endometriosis syndrome (TES). The first line therapy for TES is medical aimed at suppressing ovarian steroid hormone production. Indications for pulmonary surgery as a second line therapeutic management of patients with TES are hormonal therapy failure, intolerable adverse effects or symptom recurrence after cessation of medical treatment. Surgery should be the preferred therapeutic option when the patient is planning future pregnancies. To date, video-assisted-thoracoscopy (VATS) represents an important surgical tool in TES patients, as it allows to carry out many procedures for diagnostic and therapeutic purposes. In patients with centrally located bronchial endometriosis, sub-segmentectomies, segmentectomies or lobectomies may be required. We present the case of a patient with TES, treated with a feasible, safe and least invasive thoracic surgical technique available nowadays in many settings: uniportal-VATS.

HIV AS A RED HERRING IN SANGUINOUS PLEURAL EFFUSION SECONDARY TO THORACIC ENDOMETRIOSIS

SESSION TITLE: Tuesday Medical Student/Resident Case Report Posters SESSION TYPE: Med Student/Res Case Rep Postr PRESENTED ON: 10/22/2019 01:00 PM - 02:00 PM INTRODUCTION: Hemothorax is most commonly due to trauma and iatrogenic causes. However, spontaneous hemothorax and bloody effusions occurring independent of trauma may be secondary to malignancy, infection, rheumatologic, or vascular diseases. We present the case of a young woman with a complex bloody effusion and a medical history that lends itself to a particularly wide differential. CASE PRESENTATION: A 30 year old HIV positive woman from Ghana disclosed two weeks of cough and dyspnea during outpatient follow up with infectious disease. She denied fever, chills, sick contacts and hemoptysis, but endorsed intermittent right flank and shoulder pain. CXR showed complete whiteout of the right lung field and she was sent to the ED. On evaluation she was neither hypoxic, tachycardic, nor tachypneic. She was afebrile and without leukocytosis, though a microcytic anemia was noted. CT-TAP showed a large right pleural effusion, collapse of the right lung, leftward mediastinal shift, as well as intraperitoneal fluid and uterine fibroids. A chest tube was placed and drained 1500ml of sanguinous fluid. Fluid studies indicated an exudative effusion with pleural/serum hematocrit of 5/26.6%, failing to meet criteria for a true hemothorax . Bacterial and mycobacterial cultures were sterile, and cytology showed only hemosiderin laden macrophages. At time of discharge the etiology of the effusion was as of yet undetermined. Outpatient OBGYN evaluation included TVUS confirming fibroids and pelvic ascites and the patient endorsed dysmenorrhea and 5 years of failed attempts to conceive, collectively highly suggestive of endometriosis. At follow up with interventional pulmonology the patient’s pleural effusion had reaccumulated and thoracentesis yielded 1600ml. Cytology and immunohistochemistry were performed and PAX8, ER, and CD10 immunostains returned positive, suggesting presence of endometrial cells in the pleural space, consistent with thoracic endometriosis. DISCUSSION: In our HIV positive patient from a TB endemic region, malignant and infectious etiologies were of particular concern. Conversely, endometriosis has a prevalence of approximately 10%, the prevalence of thoracic endometriosis syndrome (TES) is uncertain but exceedingly rare, and only 14% of TES cases involve hemothorax or sanguinous effusion independent of pneumothorax. Additionally, the typical presenting symptoms of TES, cough, dyspnea, and chest pain are nonspecific. However, right sided laterality of effusion and concomitant pelvic pathology is where TES begins to stand out amongst the differentials. As with all manifestations of TES, hemothorax or bloody effusion has a predilection for right sided laterality greater than 80% of the time, and virtually all cases have concomitant pelvic involvement. CONCLUSIONS: In women of childbearing age with spontaneous hemothorax or bloody effusion, TES is a rare albeit important differential for consideration. Reference #1: Patrini D, Panagiotopoulos N, Pararajasingham J, Gvinidanidze L, Iqbal Y, Lawrence DR. Etiology and management of spontaneous haemothorax. Journal of Thoracic Disease. 2015;7(3):520-6. Reference #2: Martinez FJ, Villanueva AG, Pickering R, Becker FS, Smith DR. Spontaneous hemothorax. Report of 6 cases and review of the literature. Medicine. 1992;71(6):354-68. Reference #3: Joseph J, Sahn S. Thoracic endometriosis syndrome: New observations from an analysis of 110 cases. The American Journal of Medicine. 1996;100(2):164-70. DISCLOSURES: No relevant relationships by Jared Mickelson, source=Web Response No relevant relationships by Randal Rovinski, source=Web Response

ENDOMETRIOSIS OF THE LUNG PRESENTING AS INNUMERABLE NODULES

ENDOMETRIOSIS OF THE LUNG PRESENTING AS INNUMERABLE NODULES

THORACIC ENDOMETRIOSIS SYNDROME PRESENTING AS RECURRENT CATAMENIAL PNEUMOTHORAX

SESSION TITLE: Tuesday Medical Student/Resident Case Report Posters SESSION TYPE: Med Student/Res Case Rep Postr PRESENTED ON: 10/22/2019 01:00 PM - 02:00 PM INTRODUCTION: Catamenial pneumothorax (CP) is a rare spontaneous pneumothorax (PTX) that occurs cyclically with menstrual periods. It is part of the spectrum of thoracic endometriosis syndrome (TES) in which ectopic endometrial tissue is implanted in thoracic structures. CASE PRESENTATION: A 36-year-old G0P0 female nurse, presented with worsening right-sided pleuritic chest pain for 6 days. She had a history of stage 4 endometriosis associated with recurrent CP. Her first episode was 4 years ago when she had a large right-sided PTX in association with biopsy proven diaphragmatic endometriosis, treated with a right middle lobe wedge resection and chemical pleurodesis. She had multiple recurrences of PTX requiring repeat pleurodesis and subsequent long-term medical therapy with leuprolide and combination oral contraceptive pills. On examination, she was hemodynamically stable with decreased breath sounds in the right lower lung field. CT chest revealed a moderate volume right PTX, a sub-pleural right lower lobe mass and pleural thickening. She underwent a video-assisted thoracoscopic surgery (VATS) with combination (mechanical and chemical) pleurodesis. Pleural biopsy was negative for endometriosis. Postoperative chest x-rays revealed a resolving pneumothorax. Three days after surgery, the patient was discharged home. DISCUSSION: CP is characterized by collapsed lung, which occurs within 3 days of menstruation onset. The pathophysiology involves deposition of focal endometrial pleural implants migrating during leakage of air from the peritoneal cavity, which subsequently break down during menstruation or hypertrophy in response to hormonal changes. Another plausible theory is vasoconstriction and bronchospasm induced by prostaglandin F2 release during menses leading to alveolar rupture and PTX. Due to the vague symptomatology and limited disease awareness, the diagnosis is often missed or delayed leading to life-threatening complications. Histologically proven thoracic endometriosis is frequently but not always associated with CP. Our patient is unique as she previously had a diaphragmatic endometriosis associated CP and later had a non-TES associated bout of CP. Recent evidence suggests that combination surgical (VATS) followed by medical (GnRH agonist) therapy effectively suppresses CP recurrence. However, our patient suffered multiple recurrences even after both modalities. A combination of mechanical and chemical pleurodesis was attempted in an effort to prevent further recurrences. CONCLUSIONS: TES remains an exceedingly rare condition and very few cases have been reported. Our case is unique given her multiple recurrences and unresponsiveness to guideline directed therapies. Physicians should keep a high level of clinical suspicion for thoracic endometriosis in non-smoking women over 31 years, with a history of pelvic endometriosis, and presenting with recurrent pneumothorax. Reference #1: Junejo SZ, Singh Lubana S, Shina SS, Tuli SS.A Case of Thoracic Endometriosis Syndrome Presenting with Recurrent Catamenial Pneumothorax. Am J Case Rep. 2018 May 17;19:573-576. DISCLOSURES: No relevant relationships by Prasanta Basak, source=Web Response No relevant relationships by Stephen Jesmajian, source=Web Response No relevant relationships by Ahmed Qavi, source=Web Response

Circulating Endometrial Cells in Women With Spontaneous Pneumothorax

The occurrence of catamenial pneumothorax (CP) is rare, and the awareness of this diagnosis among physicians is insufficient. CP is highly correlated with pelvic endometriosis and remains the most common form of thoracic endometriosis syndrome. Circulating endometrial cells (CECs) have been previously detected in patients with pelvic endometriosis. Could CECs bring new insights into pneumothorax management? This study aims to describe the occurrence and molecular characteristics of CECs in women with spontaneous pneumothorax (SP) (N= 20) with high suspicion of its catamenial character. CECs were enriched from peripheral blood by size-based separation (MetaCell). In addition to cytomorphology, gene expression profiling of captured cells was performed for 24 endometriosis-associated genes. CECs were present in all 20 patients with SP. Enriched CECs exhibited four character features: epithelial, stem cell-like, stroma-like, and glandular. However, not all of them were present in every sampling. Gene expression profiling revealed two distinct phenotypes of CECs in SP and/or CP: one of them refers to the diaphragm openings syndrome and the other to endometrial tissue pleural implantations. Comparisons of the gene expression profiles of CECs in pneumothorax (CECs-SP group) with CECs in pelvic endometriosis (CECs-non-SP group) have revealed significantly higher expression of HER2 in the CECs-SP group compared with the CECs-non-SP group. This proof-of-concept study demonstrates successful isolation and characterization of CECs in patients with SP. Identification of CECs in SP could alert endometriosis involvement and help early referral to gynecologic consultation for further examination and treatment.

Recurrent pleural effusion and pain in the shoulder in women of reproductive age could have a gynecological cause: case series

Abstract Background Endometriosis is a benign disorder defined as the presence of endometrial glands and stroma outside of the uterine cavity. It has been estimated that approximately 10% of the women of reproductive age and 30–50% of the women with infertility are affected. Case presentation We report two nulliparous patients with recurrent pleural effusion and pain in the right shoulder highly suspicious for thoracic endometriosis syndrome (TES). Conclusion The objective of this study was to present patients with unusual symptoms related to gynecological disease. The second aim was to show our single institution experience as a second level hospital in terms of treatment and diagnosis of TES and diaphragmatic endometriosis.

Establishment and Characterization of a Stromal Cell Line Derived From a Patient With Thoracic Endometriosis.

Thoracic endometriosis (TE) syndrome is a clinical condition known as an extrapelvic form of endometriosis with the presence of functioning endometrial tissue involving lung parenchyma, pleura, chest wall, or diaphragm. In an effort to obtain an endometriosis ex vivo model, we established the spontaneously growing TH-EM1 cell line from endometriotic implants in lung parenchyma from a woman with TE. Maintained in long-term culture, the cells grew as large mesenchymal-like cells with a doubling time between 5 and 6 days. Treatment with medroxyprogesterone acetate (10-7 mol/L) inhibited the TH-EM1 cells growth and induced morphological changes to an epithelial-like cells. Strong expression of the nuclear estrogen receptors, progesterone receptors, and erytropoietin receptors were found in both the pulmonary implant and the TH-EM1 cells by immunohistochemical analysis. Consistent immunoreactivity of TH-EM1 cells for CD9, CD13, CD73, CD90, CD105, and CD157 was revealed by flow cytometry. Likewise, the embryonic markers, SRY-box 2 (SOX-2) and the Nanog molecules, were detected in 76% and 52% of the cells, while fetal hemoglobin and α-globin were detected in 76% and 65% of TH-EM1 cells, respectively. By RHG banding, normal metaphases were observed, while the microarray chromosomal analysis showed gains of DNA sequences located on the segments 8p23.1, 11p15.5, and 12p11.23. The described in vitro cellular model can serve as a useful tool to study the pathogenesis of endometriosis and to improve the knowledge of molecular mechanisms controlling the endometriotic cell dissemination potential.

A Case of Thoracic Endometriosis Syndrome during Dienogest Treatment

A Case of Thoracic Endometriosis Syndrome during Dienogest Treatment

Thoracic Endometriosis Syndrome: A Review of Diagnosis and Management.

Background:Endometriosis is characterized by the presence of endometrial-like glands and stroma outside the uterine cavity and is believed to affect 6%–10% of reproductive-age women. Endometriosis within the lung parenchyma or on the diaphragm and pleural surfaces produces a range of clinical and radiological manifestations. This includes catamenial pneumothorax, hemothorax, hemoptysis, and pulmonary nodules, resulting in an entity known as thoracic endometriosis syndrome (TES).Database:Computerized searches of MEDLINE and PubMed were conducted using the key words “thoracic endometriosis,” “catamenial pneumothorax,” “catamenial hemothorax,” and “catamenial hemoptysis.” References from identified sources were manually searched to allow for a thorough review.Conclusion:TES can produce incapacitating symptoms for some patients. Symptoms of TES are nonspecific, so a high degree of clinical suspicion is warranted. Medical management represents the first-line treatment approach. When this fails or is contraindicated, definitive surgical treatment for cases of suspected TES uses a combined video laparoscopy performed by a gynecologic surgeon and video-assisted thoracoscopic surgery performed by a thoracic surgeon. Postoperative hormonal suppression may further reduce disease recurrence.

Thoracic endometriosis syndrome

A 35-year-old nulliparous patient with secondary infertility presented to a tertiary hospital in Johannesburg with abnormal uterine bleeding, and a history of two previous right-sided haemopneumothoraces. The second episode had been related to her menstruation, and required the insertion of an intercostal drain in order to treat the large haemothorax. A pleural biopsy revealed pleural endometriosis. She was treated with dienogest (Visanne) on discharge, and was awaiting follow-up with the cardiothoracic surgeons for continued and definitive management.

Operative and hormonal management of recurrent catamenial pneumothorax: a case report

Catamenial pneumothorax refers to the recurrent collapse of a lung in conjunction with the menstrual cycle. It is a common manifestation of thoracic endometriosis syndrome, a rare condition involving extra-pelvic endometrial lesions. We describe a case of catamenial pneumothorax that has not recurred after treatment with hysterectomy and bilateral salpingo-oophorectomy. A 37-year-old woman with abdominal endometriosis presented with five episodes of right pneumothorax, all of which coincided with the patient’s menstrual cycle. The diagnosis of catamenial pneumothorax was made, and the patient was referred for hysterectomy and bilateral salpingo-oophorectomy. Combined transdermal estrogen/progesterone treatment was also initiated to manage her symptoms of hot flashes, vaginal dryness, and dyspareunia. Since treatment, the patient has not had recurrent pneumothorax. Due to the rarity and acuity of catamenial pneumothoraces, management options are challenging. Recommendations on the duration of combined estrogen/progesterone treatment following bilateral oophorectomy are needed. A multidisciplinary team approach consisting of pulmonologists, thoracic surgeons, and gynecologists is essential for accurate diagnosis, optimal treatment, and successful outcomes in this rare but serious condition.

A Rare Case of Cyclical Hemothorax: Thoracic Endometriosis Syndrome

Endometriosis is a common condition in which endometrial cells and stroma are deposited in extrauterine sites. Its prevalence has been estimated to be 10% of reproductive age females. It is commonly found in the pelvis; however, it may be found in the abdomen, thorax, brain, or skin. Thoracic involvement is a relatively rare presentation of this common disease. Thoracic endometriosis commonly presents as pneumothorax in 73% of patients. A rarer presentation of thoracic endometriosis is hemothorax (<14%) or hemoptysis (7%). Thoracic endometriosis is an uncommon cause of a pleural effusion. We present a case of 28-year-old African American female with no other medical conditions. She presented to the hospital with worsening right-sided pleuritic chest pain, dyspnea, and menorrhagia. She had been complaining of pleuritic chest pain for 5 years, the onset of which corresponds to the start of her menstrual cycle and is relieved with cessation of menses. Initial laboratory studies revealed a severe microcytic anemia with normal coagulation profile. Chest X-ray showed small right pleural effusion and suspicious for airspace disease. A computed tomography (CT) of chest was ordered for further clarification and identified large right pleural effusion. CT-guided thoracentesis removed 500 ml of serosanguinous fluid consisting of blood elements. There can be multiple sites involved with endometriosis and can present with wide range of symptoms that occur periodically with menses in young woman. The history and pleural fluid findings of this case are suggestive of Thoracic Endometriosis Syndrome. The diagnosis of this is often missed or delayed by clinicians, which can result in recurrent hospitalization and other complications. As internists we should be suspicious of atypical presentations of endometriosis and treat them early before complications develop. This case also highlights the importance of suspecting atypical etiologies for pleural effusion.

Thoracic and diaphragmatic endometriosis: Single-institution experience using novel, broadened diagnostic criteria

Objective:To describe our experience with the multidisciplinary management of both thoracic/diaphragmatic endometriosis (TED), applying a broadened definition of the “Thoracic endometriosis syndrome (TES)” to define cases.Material and Methods:We present a retrospective series of consecutive patients affected by pathology-proven TED, treated at our institution, during a period of 7 years.Results:Five women were included. Two patients were referred due to catamenial chest/shoulder pain, one due to recurrent catamenial pneumothorax, and one due to new-onset diaphragmatic hernia. One patient had no thoracic symptoms, but diaphragmatic endometriosis was found during gynecologic laparoscopy for pelvic endometriosis. Endometriosis was histologically confirmed in all cases. After follow-up, all patients remain asymptomatic.Conclusion:Broadened TES criteria could increase the incidence of TED and determine better knowledge of this condition. Multidisciplinary, minimally invasive surgery is effective and safe, but should be reserved for tertiary referral centers.

An Asymptomatic Haemorrhagic Pleural Effusion Mimicking Extra-Pulmonary Tuberculosis in a Young Woman with Thoracic Endometriosis Syndrome: A Case Report

A young woman presented with an asymptomatic recurrent right sided pleural effusion. Pleural fluid full report showed neutrophilic and haemorrhagic effusion. Adenosine deaminase level in the pleural fluid was elevated and the Mantoux test was positive. Microbiological diagnosis was negative, and she was started on anti-tuberculous therapy on clinical grounds for extrapulmonary tuberculosis. Diagnostic Thoracoscopy revealed characteristic appearance suggestive of thoracic endometriosis which was confirmed by histology.

Thoracic endometriosis syndrome at University of Ilorin Teaching Hospital.

Background Endometriosis is defined as the presence of endometrial tissue (stroma and functional glands) outside the uterine cavity in women of reproductive age. Ectopic sites are frequently located in the pelvis; extrapelvic sites have been reported in the gastrointestinal tract and thoracic cavity. Thoracic manifestation of endometriosis constitutes thoracic endometriosis syndrome (TES). Objectives To examine the presentation pattern and outcome of in the management of TES. Methods This study is a retrospective review of medical records of patients diagnosed with endometriosis at the University of Ilorin Teaching Hospital over a 3.5-year period from January 2014 to June 2017. Results A total of 21 patients presented with endometriosis, of whom 8 (38.1%) presented with TES. The most common variety of TES was catamenial pleural effusion (CPE) accounting for 75%, followed by catamenial chest pain (37.5%). Two patients (25%) each presented with catamenial pneumothorax and catamenial haemoptysis, while 1 (12.5%) had catamenial surgical emphysema. Closed thoracostomy tube drainage plus chemical pleurodesis was the most frequent intervention technique, accounting for 62.5%. Conclusion TES remains an uncommon entity, despite being the most common extrapelvic manifestation of endometriosis. CPE appeared to be the most common variant of TES in our environment. Currently available treatment options need to be improved, and more use made of video-assisted thoracoscopic surgery.

Thoracic endometriosis syndrome at the University of Ilorin Teaching Hospital

Thoracic endometriosis syndrome at the University of Ilorin Teaching Hospital

Thoracic Endometriosis

Abstract: A recurrent hemorrhagic pleural effusion in a woman during her reproductive years may be the clinical presentation of thoracic endometriosis syndrome (TES) . We report here a case of a recurrent bloody right pleural effusion in a young female who had a history of pelvic endometriosis. Thoracic endometriosis with pleural involvement was confirmed by pleural biopsy which showed focal involvement of functional endometrial tissue within the pleura. The patient underwent pleurectomy and talc pleurodesis without recurrence of the pleural fluid. A hemorrhagic pleural effusion due to thoracic endometriosis may mimic other conditions including pulmonary thromboembolism, trauma, malignancy, tuberculosis, and others. Thoracic endometriosis should be in the differential diagnosis of a bloody pleural effusion in women of childbearing age, particularly in a patient with an established diagnosis of pelvic endometriosis.

Thoracic endometriosis syndrome: Comparison between catamenial pneumothorax or endometriosis-related pneumothorax and catamenial hemoptysis

ObjectivesTo evaluate the clinical features of thoracic endometriosis syndrome (TES) represented by catamenial pneumothorax (CP), endometriosis-related pneumothorax (ERP), and catamenial hemoptysis (CH). Study designIn this retrospective study, we enrolled 25 patients with TES, 18 of whom had CP/ERP and 7 had CH, to investigate the clinical presentation, effectiveness of treatment, and recurrence rates in these disorders. ResultsThe age at onset was significantly lower in patients with CH than in patients with CP/ERP (P < 0.05). In 94.4% of patients with CP/ERP, pneumothorax was observed on either the right side or bilaterally, however there was no tendency toward laterality of CH among our cases. In our study, patients with CP/ERP predominantly underwent surgical management and the recurrence rate during treatment was higher in patients with CP/ERP than in those with CH. We found that the recurrence frequency of CP/ERP was lowest under the combination therapy with thoracic surgery and postoperative hormonal therapy. ConclusionOur findings suggest that CP/ERP and CH are different pathological conditions and CP/ERP is more difficult to manage than CH.