{"paper_id":"2580c26d-57d9-4311-9f8f-dd9b2cb597f5","body_text":"Submit Manuscript | http://medcraveonline.com\nAbbreviations: CPP, chronic pelvic pain; MIGS, invasive \ngynecologic surgery; TES, thoracic endometriosis syndrome; \nV ATS, video-assisted laparoscopy surgery; SILS, single-incision \nlaparoscopic surgery; OCPs, oral contraceptive pills; MRI, Magnetic \nresonance imaging; GnRH, gonadotropin-releasing hormone; BSO, \nbilateral salpingo-oophorectomy\nBackground\nCyclic chest pain with a catamenial relationship should increase \nsuspicion of thoracic involvement with endometriosis. Similar to \npelvic lesions these lesions are amenable to surgical resection. Due to \nthe uncommon nature of thoracic involvement guidance and support \nfrom a gynecologic surgeon is a valuable resource to ensure complete \nand correct excision of suspected lesions. \nPresentation of the case\nPatient is a 29-year-old Gravida 2, Para 0020 with known history \nof endometriosis, who was referred to our clinic for further evaluation. \nThe patient complained of chronic pelvic pain, endometriosis, \ncatamenial hemoptysis (spitting blood with menses), cyclic chest \npain, dysmenorrhea, and menorrhagia. \nThe patient’s surgical history was significant for previous \nlaparoscopic excisions/resection of endometriosis performed 2 years \nbefore presentation. Her family history was also significant for \nendometriosis.\nOn pelvic examination, there is bilateral adnexal tenderness with \nposterior fornix sweeping pain. On respiratory examination, there \nis catamenial shortness of breath and right sided chest pain with no \nhemoptysis. \nChest CT was normal with no visible lung nodules, implants, \nor pneumothoraxes. Transvaginal and transabdominal ultrasound \nshowed an endometrioma or a hemorrhagic cyst within the right ovary \nmeasuring 3.4x3.7cm with the right ovary measuring 4.2x4.4cm.\nThe patient underwent robotic single-incision laparoscopic \nsurgery (SILS) resection of endometriosis, ovarian cystectomy, lysis \nof adhesions, and cystoscopy by Minimally Invasive Gynecologic \nSurgery (MIGS) team. After completion of the patient’s pelvic \nsurgery, the thoracic surgery team performed bilateral video-assisted \nlaparoscopy surgery (V ATS) with assistance from the MIGS to \nhelp identify suspicious lesions. Intraoperative evaluation revealed \nthoracic endometriosis confined to the pleura of the lungs and the \nObstet Gynecol Int J. 2019;10(5):342‒346. 342\n©2019 Rezai et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which \npermits unrestricted use, distribution, and build upon your work non-commercially.\nThoracic endometriosis, a review\nVolume 10 Issue 5 - 2019\nShadi Rezai,1 Alexander G Graves,2 \nCassandra E Henderson,3 Xiaoming Guan1\n1Division of Minimally Invasive Gynecologic Surgery, \nDepartment of Obstetrics and Gynecology, Baylor College of \nMedicine, USA\n2University of Queensland, Mayne Medical School, Australia\n3OB/GYN At Lake Success, One Hollow Lane, USA\nCorrespondence: Xiaoming Guan MD PhD, Division \nChief and Fellowship Director, Division of Minimally Invasive \nGynecologic Surgery, Department of Obstetrics and \nGynecology, Baylor College of Medicine, 6651 Main Street, 10th \nFloor, Houston, T exas, 77030, USA, T el (832) 826-7464, Fax (832) \n825-9349, Email \nReceived: July 21, 2019 | Published: September 24, 2019\nAbstract\nBackground: Thoracic endometriosis is the most frequent extra-pelvic location of \nendometrial lesions. Because thoracic endometriosis is an unusual and uncommon \ndiagnosis in women, it is crucial that patients with catamenial chest pain and previous \nhistory of endometriosis undergo a thorough work-up. Due to the rarity of this disease \na high index of clinical suspicion is imperative to make a diagnosis. Consequently, due \nto the multi-organ involvement of this disease a multi-disciplinary team is required for \nappropriate investigation, diagnosis, and treatment. \nPresentation of the case: Patient is a 29-year-old Gravida 2, Para 0020 with a known \nhistory of pelvic endometriosis, confirmed by histopathology, was referred to our clinic for \nevaluation of her chronic pelvic pain, endometriosis, catamenial dyspnea, cyclic chest pain, \ndysmenorrhea, and menorrhagia. \nThe patient underwent robotic single-incision laparoscopic surgery (SILS) resection of \nendometriosis, ovarian cystectomy, lysis of adhesions, and cystoscopy by the Minimally \nInvasive Gynecologic Surgery (MIGS) team. Afterwards, the thoracic surgery team \nperformed bilateral video-assisted laparoscopy surgery (V ATS) with assistance from the \nMIGS to help identify suspicious lesions. Intraoperative evaluation revealed thoracic \nendometriosis confined to the pleura of the lungs and the diaphragm and were resected. \nBased on the clinical presentation and intraoperative findings the patient was diagnosed \nwith thoracic endometriosis.\nConclusion: Thoracic endometriosis is an unusual, but relatively common diagnosis in \nwomen with catamenial chest pain, catamenial dyspnea, and previous history of confirmed \nendometriosis. Due to the impacts on patient quality of life and the potential complications \nof thoracic endometriosis, we find it imperative to investigate this patient population. \nKeywords: catamenial hemoptysis, catamenial Pneumothorax, chronic pelvic pain, \ncatamenial symptoms, cyclic chest pain, diaphragmatic endometriosis, Dienogest, \ndyspnea, elagolix (orilissa), endometriosis, endometriosis complications, endometriosis \nrelated pneumothorax, endometriosis surgery, minimally invasive gynecologic surgery, \nmultidisciplinary team, orilissa, thoracic endometriosis, thoracic endometriosis syndrome, \nvideo-assisted laparoscopy surgery\nObstetrics & Gynecology International Journal\nCase Report\n Open Access\n\n\nThoracic endometriosis, a review\n343\nCopyright:\n©2019 Rezai et al.\nCitation: Rezai S, Graves AG, Henderson CE, et al. Thoracic endometriosis, a review. Obstet Gynecol Int J. 2019;10(5):342‒346. \nDOI: 10.15406/ogij.2019.10.00464\ndiaphragm. These lesions were resected via electrocautery (Figures \n1–3). No parenchymal lung lesions were identified. The patient had \nan uncomplicated recovery and postoperative phase. Upon discharge \nthe patient was started on oral contraceptive pills (OCPs). Based on \nthe clinical presentation and intraoperative findings the patient was \ndiagnosed with thoracic endometriosis.\nFigure 1 Endometriosis of the right ovary.\nFigure 2 Lesions on the right pleural surface.\nFigure 3 Lesions on the central tendon of the diaphragm (Diaphragmatic \nEndometriosis).\nDiscussion\nEndometriosis is a benign condition characterized by uterine \nendometrial tissue outside of its normal location—most commonly \ninvolving the ovaries. 1 In patients with extra-pelvic manifestations, \nthoracic involvement is most common, especially in patients with \ncurrent pelvic disease. 2,3 Laparoscopic resection is the gold standard \nfor pelvic endometriosis, 4 while evidence for surgical resection of \nthoracic endometriosis is limited due to the rarity of the disease. 5 \nTherefore, coordinated care is of the utmost importance when treating \nthis disease when it appears outside the pelvic cavity.\nWhen discussing management of thoracic endometriosis there are \nimportant terms to define6:\na. Thoracic endometriosis—endometrial tissue found in the thorax \nthat is identified on histological specimens (hormone receptor-\npositive endometrial stroma and glands) obtained from chest \ntube aspirate, thoracotomy, or bronchoscopy.\nb. Probable thoracic endometriosis—the identification of tissue \nwithin the thorax that is suggestive but not definitively diagnostic \nof endometrium (e.g., stroma only or hormone receptor-negative \ntissue).\nc. Thoracic endometriosis syndrome—one or more clinical \nmanifestations of thoracic involvement is present (e.g., \npneumothorax, hemothorax, hemoptysis, chest pain) in \nassociation with menstruation but without histological \nconfirmation.\nd. Catamenial—the occurrence of symptoms or signs that bear a \ntemporal relationship with menses.\nThere are several proposed mechanisms on how these lesions \ndevelop in the thorax. The three most popular theories are auto \ntransplantation via retrograde menstruation, micro-embolization/\nmetastasis, and coelomic metaplasia with auto transplantation being \nthe most popular theory.7\nThe autotransplantation via retrograde menstruation theory \nproposes that ectopic endometrium may migrate through \ndiaphragmatic defects. 8,9 However, three different observations \nweaken this theory. First, diaphragmatic defects are uncommon, \nand the overwhelming majority of thoracic endometriosis are \npredominantly right-sided (~95% of cases);  3  if defects were truly \nportals of entry for endometrial tissue, the disease would be expected \nto be bilateral in distribution. Second, there can be a recurrence of \nsymptoms even after a diaphragmatic repair and/or hysterectomy. 10 \nFinally, ectopic endometrial tissue differs significantly from eutopic \nendometrium in clonality, enzymatic activity, protein expression, and \nhistologic properties.11,12\nThe micro-embolization/metastasis theory suggests metastatic \nspread of endometrial tissue through the venous or the lymphatic \nsystem to the lungs. 13,14 Endometrial foci in tissue remote from the \npelvis and thorax (e.g., brain, knee, and eye) support the “metastatic \ntheory.” However, in opposition to this theory, micro-embolization \nwould be expected to affect both hemithoraces approximately equally, \nwhereas thoracic endometriosis is found overwhelmingly in the right \nhemothorax.14\nThe coelomic metaplasia theory suggests there is a transformation \nof pluripotent cells to differentiated endometrium. In support of this \ntheory, pluripotent cells have been identified in uterine endometrium \n\n\nThoracic endometriosis, a review\n344\nCopyright:\n©2019 Rezai et al.\nCitation: Rezai S, Graves AG, Henderson CE, et al. Thoracic endometriosis, a review. Obstet Gynecol Int J. 2019;10(5):342‒346. \nDOI: 10.15406/ogij.2019.10.00464\nand a case of endometriosis has been reported in a 20-year-old woman \nwith congenital agenesis of uterus, cervix, vagina, and fallopian \ntubes.15\nThe etiology is likely to be multifactorial given that not one \ntheory explains the pathogenesis of endometriosis, but rather a \nconglomeration of the three theories does.\nClinical presentation\nThere are four main clinical presentations with thoracic \nendometriosis: catamenial pneumothorax, catamenial hemothorax, \ncatamenial hemoptysis, and pulmonary nodules. The most common \npresentation (70 to 73%) of thoracic endometriosis is catamenial \nPneumothorax.5 A smaller proportion (approximately 15%), has \ncatamenial pneumothorax due to probable thoracic involvement, \nand 10 percent or less have pneumothorax that has no temporal \nrelationship with menses (non-catamenial pneumothorax). 5 Patients \nwith endometriosis-related non-catamenial pneumothorax are \ntypically identified during surgery for recurrent pneumothorax. \nCatamenial hemothorax is the next most common manifestation \nof thoracic endometriosis. Hemorrhage into the pleural space occurs \nin less than 15 percent of patients with thoracic endometriosis. 3,14 \nCompared with endometriosis-associated pneumothorax, \nendometriosis-associated hemothorax is typically associated with \nextensive pleural endometrial implants.\nCatamenial hemoptysis occurs in less than 14 percent of patients \nand is typically due to parenchymal or endobronchial endometriosis.3,16 \nThe mean age of patients presenting with hemoptysis (26 to 29 \nyears) appears to be lower than patients with other manifestations of \nthoracic endometriosis (34 years). 3,17 Bleeding is usually catamenial, \nminor, and right-sided. The least common manifestation of thoracic \nendometriosis is pulmonary nodules. It can present as solitary or \nmultiple pulmonary nodules (2 to 6% of all patients). Nodules can \nbe incidental or symptomatic (e.g., hemoptysis or chest pain), vary in \nsize (0.5 to 3cm) and are typically right-sided.3,18,19\nIn patients with early disease that is minimally and mainly \nconfined to the diaphragm the above four clinical presentations may \nnot be appreciable In cases where the endometriosis is confined to \nthe diaphragm there can be associated pain in the ipsilateral chest, \nshoulder, arm, and neck and rarely can lead to diaphragmatic rupture \nand pneumoperitoneum.20\nDiagnosis\nAs there is no gold standard diagnostic method, test, or imaging \nfor thoracic endometriosis the diagnosis is usually on a clinical \nground. Since this is a rare disease and there is a wide spectrum of \npresentation, a high index of clinical suspicion is key to recognize \nthis disease. When a premenopausal woman presents with catamenial \ndyspnea, cough, and chest pain it is crucial the examining physician \nassess the possibility of thoracic involvement of endometriosis and \nmake it component of the differential diagnosis. Symptoms have a \ncatamenial pattern, occurring between 24 hours before and 72 hours \nafter the onset of menses, 18 and typically recurring. 5 Catamenial \npneumothorax is defined by at least two episodes of pneumothorax \noccurring during this time interval. 5 The right-side predominance \nof symptoms represents a diagnostic clue. A thorough and targeted \nphysical examination may reveal diminished or absent breath \nsounds, dullness to percussion and decreased chest expansion, which \ncould suggest a pleural effusion. Furthermore, decreased breath \nsounds, hyperresonance, and decrease chest expansion suggest a \npneumothorax. \nImaging is important to evaluate any history and focal exam \nfindings suggesting thoracic pathology. A chest x-ray can help \nelucidate a pneumothorax, pleural effusions, or pulmonary nodules. \nCT can further assess if there is a pneumothorax, pleural effusions, or \npulmonary nodule if the chest x-ray was inconclusive. However, CT \nis considered the first-line imagining, as it can rule out other diagnoses \nand map the lesions for surgery if necessary. 21 Unfortunately, CT \nimagining is poorly specific; therefore, its main role is to rule out \nother pulmonary diseases. \nMagnetic resonance imaging (MRI) is an alternative to CT imaging. \nThe lack of spatial resolution, as compared to CT, is compensated \nby a higher contrast resolution and allows a better depiction of \nhemorrhagic lesions. There are two drawbacks with the use of MRI. \nFirst, a single breath hold is easily achievable with a CT, but this \nbecomes an impossible feat with the MRI. Second, the presence of air \nclose to the diaphragm may make it difficult to distinguish true lesions \nfrom artefacts. Overall, MRI may be superior to CT only because of \nless radiation exposure.22\nThere is a limited role for bronchoscopy in diagnosis because most \npathologic features are located in the peripheral lung. There have been \ncase reports of success with bronchoscopy, but there are few cases \npresented in the literature.23\nTreatment\nDespite the lack of randomized studies, there is disagreement \nin the literature regarding first line therapy as whether it should be \nsurgical or medical therapy first. Some experts believe that the \nmajority patients with thoracic endometriosis should be treated with \nhormone suppressive therapy as opposed to surgery as first line \ntherapy. Data from observational studies support a reduction in the \nrate of recurrent pneumothorax and hemothorax with hormonal agents \nalone.24 Whereas, this is support for surgical management as first line \ntherapy when there is no/low response to hormonal therapy.25\nIn regard to medical therapy, Dienogest, a progestogenic drug with \nmoderate estrogen suppression, anti-inflammatory, antiproliferative, \nand anti-angiogenic properties, is a new treatment available for initial \nmanagement of thoracic endometriosis.17\nTypically, gonadotropin-releasing hormone (GnRH) analogs are \nthe first-line agents because they are highly effective at suppressing \novarian hormone production and inhibiting the growth of endometrial \ntissue.3,5,26,27 However, prolonged therapy with GnRH analogs is often \ncomplicated by intolerance from the acute symptoms of menopause \nand the risk of osteoporosis. Consequently, the decision to use GnRH \nanalogs is dependent upon weighing the benefits of reduced recurrence \nagainst the adverse effects in the context of patient preferences \nregarding the temporary induction of menopause and inability to \nbecome pregnant.\nFor those not willing to become menopausal or who have a \ncontraindication to GnRH analogs, oral contraceptives or progestins, \nand less commonly, danazol or aromatase inhibitors, are alternatives. \nThe advantage of oral contraceptive agents is that endometrial implant \nstimulation is low grade and withdrawal bleeding can be avoided, if \nnecessary, by the administration of continuous therapy. All hormonal \nagents should be stopped in those who cannot tolerate the side effects \nand in those who have a desire to become pregnant. Recurrence \n\nThoracic endometriosis, a review\n345\nCopyright:\n©2019 Rezai et al.\nCitation: Rezai S, Graves AG, Henderson CE, et al. Thoracic endometriosis, a review. Obstet Gynecol Int J. 2019;10(5):342‒346. \nDOI: 10.15406/ogij.2019.10.00464\nrates are high particularly when hormonal suppressive therapy is not \nadministered. However, some patients develop recurrence despite use \nof hormonal therapy.\nIn patients who are unable to undergo medical management or \nwho have refractory disease, surgery may be a secondary treatment \noption. However, the optimal therapy for patients with refractory \ndisease is unknown. \nIf the patient and surgeon agree on surgery as first line, current \navailable options are: pleurectomy and repeat pleurodesis for \nrecalcitrant pneumothorax or hemothorax and hysterectomy with \nor without bilateral salpingo-oophorectomy (BSO). In the thorax, \nall surgical techniques should be directed at pleural adhesion to the \nthoracic wall to remove any potential space for pathologic features. 28 \nAlthough, these surgical options cause infertility, surgical management \nshould always be discussed.\nOnce the patient has been treated for thoracic endometriosis \nwith or without pelvic involvement, she should be followed by her \ngynecologist as well as her pulmonary surgeon at regular intervals \nto assess reoccurrence until it is deemed, she has reached a time of \ndisease-free progression.\nConclusion\nDue to the numerous anatomic locations of endometriosis and \nthe inherent complexity of the disease we find it is imperative that \na multi-disciplinary team delivers coordinated care to achieve the \nbest outcome for the patient. We have found the two-team approach \nto be the safest and most effective approach to treating patients \nwith thoracic endometriosis, consisting of a thoracic surgeon and \ngynecologist. While the thoracic surgeon is operating in the thorax, \nthe gynecologist is present to assist and advise on any suspicious \nlesions that may require resection. \nIn terms of diagnosis, specialists must have a high index of \nsuspicion for thoracic endometriosis when a patient presents with \ncatamenial chest symptoms on a background of pelvic endometriosis. \nIn these patients, further history, focused examination, and \ninvestigations should be initiated, including chest imaging (CT or \nMRI) to rule out another chest pathology. Video assisted thoracic \nsurgery (V ATS) should also be discussed with the patient as a mode \nof definitive diagnosis. \nFirst line treatment of thoracic endometriosis is still debated within \nthe literature,. Ultimately, we find that the direction of therapy, whether \nit be non-invasive or invasive measures, is strongly guided by patient \npreference. As we see patients in our surgical gynecology clinic, the \nvast majority of them have failed medical hormonal therapy or it has \nnot been sufficient to reduce their symptoms. We offer minimally \nsurgery to these patients as a more definitive treatment. The risks \nand benefits are thoroughly discussed with the patient with adequate \ninsight into how each treatment option can affect their personal lives \nand their reproductive health. \nAcknowledgments \nDr. Xiaoming Guan is a speaker for Applied Medical, Rancho \nSanta Margarita, California.\nFunding\nNone.\nConflicts of interest\nAuthors did not report any potential conflicts of interests.\nReferences\n1. Giudice LC, Kao LC, Endometriosis. Lancet. 2004;364(9447):1789–1799. \n2. Bagan P, Berna P, Assouad J, et al. Value of cancer antigen 125 for diagnosis \nof pleural endometriosis in females with recurrent pneumothorax. Eur \nRespir J. 2008;31(1):140–142. \n3. Joseph J, Sahn SA. 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