{"paper_id":"61bd6fa2-fffb-4b05-a4fe-3ea59d02834b","body_text":"Review began\n 09/27/2024 \nReview ended\n 10/07/2024 \nPublished\n 10/10/2024\n© Copyright \n2024\nÁvila et al. This is an open access article\ndistributed under the terms of the Creative\nCommons Attribution License CC-BY 4.0.,\nwhich permits unrestricted use, distribution,\nand reproduction in any medium, provided\nthe original author and source are credited.\nDOI:\n 10.7759/cureus.71202\nHemopneumothorax Secondary to Pleural\nEndometriosis in a Woman of Childbearing Age:\nA Rare Presentation\nLeonor Ávila \n, \nHelena Leandro \n, \nRita Silva \n, \nRui Mendes \n, \nFátima Coelho \n1.\n General Surgery, Unidade Local de Saúde Lisboa Ocidental, Lisbon, PRT\nCorresponding author: \nLeonor Ávila, \nleonorcostasantos@gmail.com\nAbstract\nPleural endometriosis is a rare condition characterized by the presence of endometrial tissue outside the\nuterus, involving the pleura. This atypical condition can be asymptomatic or cause respiratory symptoms\nsuch as chest pain, dyspnea, and hemothorax. We present a clinical case of a 32-year-old woman who\npresented to the emergency room with hemothorax due to pleural endometriosis.\nCategories:\n Emergency Medicine, Obstetrics/Gynecology, General Surgery\nKeywords:\n chest drainage, endometriosis, hemopneumothorax, hemothorax, pleural endometriosis, thoracic\nendometriosis\nIntroduction\nEndometriosis is a condition characterized by the presence of ectopic endometrial tissue, commonly within\nthe pelvic region, but can occur in extrapelvic sites, including the pleural cavity, bowel, brain, diaphragm,\nand skin \n[1]\n. Endometriosis is a non-cancerous, inflammatory condition influenced by estrogen that impacts\nfemales during their premenstrual, reproductive, and postmenopausal phases.\nThoracic endometriosis is an uncommon condition that mainly affects young women. Its prevalence in the\ngeneral population is not well documented, but it has been observed in less than 1% of women undergoing\npelvic surgery for suspected or confirmed pelvic endometriosis \n[2, 3]\n. Higher rates are seen in individuals\nwith primary spontaneous pneumothorax, ranging from 3% to 6%. For those with recurrent pneumothorax\nor cases requiring surgery, the rates increase to between 6% and 20%, while in cases of catamenial\npneumothorax, the rates can be as high as 65% to 89%. Depending on its manifestation, it can be\nasymptomatic or cause severe respiratory symptoms, which can be life-threatening \n[4-8]\n. \nIn this report, we present a clinical case involving a 32-year-old woman who arrived at the emergency room\nwith hemothorax, caused by pleural endometriosis, and required prompt attention and medical care. This\ncase emphasizes the challenges and vital significance of timely diagnosis, as well as the necessity for\nproactive management to reduce the risk of serious complications associated with this condition.\nThe patient provided informed consent for the use of her clinical information and medical photographs, and\nher privacy and confidentiality were strictly maintained throughout the process.\nCase Presentation\nA 32-year-old nulliparous woman with a known history of endometriosis diagnosed by previous laparoscopy\nwith excision of endometriosis' implants on the peritoneal cavity, presented to the emergency department\nwith acute onset right chest pain, dyspnea, and a history of recent menstruation. She reported a recurrent\nright-sided pleuritic plain that had worsened over the past 48 hours. The patient denied any history of\ntrauma or other significant medical conditions. Physical examination revealed decreased breath sounds on\nthe right side and dullness to percussion. A chest X-ray and computed tomography (CT) scan demonstrated a\nlarge right-sided hemothorax. Hemodynamic instability with tachycardia (heart rate (HR) 110 beats per\nminute (bpm)) and hypotension (85/70 mmHg) was noted. Still, normal values of oxygen saturation (96%)\nand partial pressure of oxygen (pO\n2\n 78.2 mmHg) were present (Figures \n1\n-\n3\n).\n1\n1\n1\n1\n1\n \nOpen Access Case Report\nHow to cite this article\nÁvila L, Leandro H, Silva R, et al. (October 10, 2024) Hemopneumothorax Secondary to Pleural Endometriosis in a Woman of Childbearing Age: A\nRare Presentation. Cureus 16(10): e71202. \nDOI 10.7759/cureus.71202\n\nFIGURE\n 1: Chest X-ray demonstrating the hemothorax\nFIGURE\n 2: Thoracic CT scan (axial plane) demonstrating the\nhemothorax (red arrow)\n \n2024 Ávila et al. Cureus 16(10): e71202. DOI 10.7759/cureus.71202\n2\n of \n6\n\nFIGURE\n 3: Thoracic CT scan (coronal plane) demonstrating the\nhemothorax (red arrow)\nLaboratory investigations showed a decreased level of hemoglobin (6 g/dL) consistent with active bleeding\ninto the pleural space; she also presented with leukocytosis (13.7 x 10\n9\n/L) and an increased level of C-\nreactive protein (6.5 mg/dL). Regarding renal function, it was evident an acute renal failure, with a serum\ncreatinine level of 1.40 mg/dL with normal urea levels. The blood coagulation parameters were within\nnormal ranges. Immediate resuscitation, with transfusion of one unit of red blood cells, and chest tube\nplacement were performed, which revealed frank blood (850 mL), and pleural fluid analysis confirmed the\npresence of endometrial cells (Figure \n4\n).\n \n2024 Ávila et al. Cureus 16(10): e71202. DOI 10.7759/cureus.71202\n3\n of \n6\n\nFIGURE\n 4: Chest X-ray demonstrating the chest tube\nThe red arrow demonstrates the chest tube in the sixth intercostal space.\nA diagnosis of hemothorax secondary to pleural endometriosis was made based on clinical presentation\n(woman of childbearing age with acute respiratory symptoms), radiological findings (evidence of\nhemothorax on radiologic exams), and the presence of endometrial cells in pleural fluid. \nHormonal therapy with a gonadotropin-releasing hormone (GnRH) agonist was initiated promptly to induce\namenorrhea and reduce endometrial tissue activity. The chest tube was removed once the hemothorax\nresolved (last output of 50 mL/day), and the patient was discharged after five days with a plan for long-term\nhormonal therapy and close follow-up, with a gynecology consult, to monitor for recurrence (Figure \n5\n).\n \n2024 Ávila et al. Cureus 16(10): e71202. DOI 10.7759/cureus.71202\n4\n of \n6\n\nFIGURE\n 5: Chest X-ray at the discharge demonstrating the resolution of\nthe hemothorax\nDiscussion\nPleural endometriosis is a rare condition that can occur in isolation or in association with pelvic\nendometriosis. Several theories have been put forward to explain how thoracic endometriosis develops,\nsome of which overlap with those for pelvic disease. They are as follows: (1) Autotransplantation via\nretrograde menstruation: Sampson's theory suggests that endometrial tissue can reach the thoracic cavity\nthrough retrograde menstruation, where menstrual blood flows backward through the fallopian tubes,\nleading to the transplantation of endometrial cells into both the peritoneal and thoracic cavities \n[9]\n; (2)\nMicro-embolization/metastasis: Another proposed mechanism is the metastatic spread of endometrial\ntissue via the venous or lymphatic systems to the lungs \n[10]\n. Evidence supporting this includes the discovery\nof endometrial foci in areas distant from the pelvis and thorax, such as the brain, knee, and eye.\nAdditionally, circulating endometrial cells have been found in 90% of patients with confirmed pelvic\nendometriosis; (3) Coelomic metaplasia: This theory posits that pluripotent cells can transform into\ndifferentiated endometrial tissue, a process known as coelomic metaplasia. Support for this idea comes from\nthe identification of pluripotent cells in the uterine endometrium \n[11, 12]\n.\nThe diagnosis of pleural endometriosis is challenging and often requires a high clinical suspicion. Clinical\nsymptoms can range from asymptomatic until pleuritic chest pain or even massive hemothorax, as observed\nin this case \n[4-8]\n.\nDetailed medical history with special attention to the menses, imaging studies that can reveal endometrial\nimplants, and pleural fluid analysis (demonstrating the presence of endometrial cells and stroma within the\npleura) are valuable tools for accurate diagnosis.\nThe management of patients with thoracic endometriosis frequently involves multiple disciplines, including\npulmonologists, thoracic surgeons (surgical resection of endometriotic implants to prevent hemothorax\nrecurrence), and gynecologists. In this particular case, after discharge, the patient was oriented to a thoracic\nsurgery appointment with the goal that she would subsequently undergo surgery to remove the thoracic\nendometriosis implants. Because of the multiple different disciplines involved, good communication among\nclinicians is critical for successful outcomes. Nevertheless, treatment choice should be individualized and\ndiscussed with the patient, considering her reproductive desires \n[8]\n.\nConclusions\nPleural endometriosis is a rare manifestation of endometriosis that can be silent or present with severe\nsymptoms such as hemothorax. While thoracic endometriosis can sometimes occur isolated, it typically\n \n2024 Ávila et al. Cureus 16(10): e71202. DOI 10.7759/cureus.71202\n5\n of \n6\n\npresents alongside significant endometriosis affecting the reproductive, genitourinary, and gastrointestinal\nsystems. In patients diagnosed with thoracic endometriosis, approximately 50% to 84% also have pelvic\nendometriosis. Early diagnosis and appropriate treatment are essential to prevent complications and ensure\nthe patient's complete recovery. This case report highlights the importance of considering pleural\nendometriosis in patients with acute respiratory symptoms in women of childbearing age, especially in the\npresence of known endometriosis, and underscores the need for a multidisciplinary approach to effective\nmanagement. Close follow-up is essential to monitor for recurrence.\nAdditional Information\nAuthor Contributions\nAll authors have reviewed the final version to be published and agreed to be accountable for all aspects of the\nwork.\nConcept and design:\n  \nLeonor Ávila, Rita Silva, Rui Mendes, Fátima Coelho\nAcquisition, analysis, or interpretation of data:\n  \nLeonor Ávila, Helena Leandro\nDrafting of the manuscript:\n  \nLeonor Ávila, Helena Leandro, Rita Silva, Rui Mendes, Fátima Coelho\nCritical review of the manuscript for important intellectual content:\n  \nLeonor Ávila, Rita Silva, Rui\nMendes, Fátima Coelho\nSupervision:\n  \nRui Mendes, Fátima Coelho\nDisclosures\nHuman subjects:\n Consent was obtained or waived by all participants in this study. \nConflicts of interest:\n In\ncompliance with the ICMJE uniform disclosure form, all authors declare the following: \nPayment/services\ninfo:\n All authors have declared that no financial support was received from any organization for the\nsubmitted work. \nFinancial relationships:\n All authors have declared that they have no financial\nrelationships at present or within the previous three years with any organizations that might have an\ninterest in the submitted work. \nOther relationships:\n All authors have declared that there are no other\nrelationships or activities that could appear to have influenced the submitted work.\nReferences\n1\n. \nTsamantioti ES, Mahdy H: \nEndometriosis\n. StatPearls [Internet], StatPearls Publishing, Treasure Island, FL;\n2024.\n2\n. \nViganò P, Parazzini F, Somigliana E, Vercellini P: \nEndometriosis: epidemiology and aetiological factors\n. Best\nPract Res Clin Obstet Gynaecol. 2004, 18:177-200. \n10.1016/j.bpobgyn.2004.01.007\n3\n. \nGuo SW, Wang Y: \nThe prevalence of endometriosis in women with chronic pelvic pain\n. Gynecol Obstet\nInvest. 2006, 62:121-30. \n10.1159/000093019\n4\n. \nPorcel JM, Sancho-Marquina P, Monteagudo P, Bielsa S: \nPleural effusion secondary to endometriosis: a\nsystematic review\n. Am J Med Sci. 2023, 366:296-304. \n10.1016/j.amjms.2023.08.003\n5\n. \nSakharuk I, Drevets P, Coffey P, Guitton J, Patel V: \nPleural endometriosis: an atypical cause of hemoptysis\n.\nAm Surg. 2023, 89:3292-4. \n10.1177/00031348231161715\n6\n. \nKyejo W, Ismail A, Rubagumya D, Bakari R, Kaguta M, Matillya N: \nShortness of breath in a young lady, rare\ncase report of thoracic endometriosis\n. Int J Surg Case Rep. 2022, 95:107226. \n10.1016/j.ijscr.2022.107226\n7\n. \nWang P, Meng Z, Li Y, Xu Z: \nEndometriosis-related pleural effusion: a case report and a PRISMA-compliant\nsystematic review\n. Front Med (Lausanne). 2021, 8:631048. \n10.3389/fmed.2021.631048\n8\n. \nErratum regarding missing declaration of competing interest and patient consent statements in previously\npublished articles\n. Radiol Case Rep. 2023, 18:1643-4. \n10.1016/j.radcr.2023.01.017\n9\n. \nVinatier D, Orazi G, Cosson M, Dufour P: \nTheories of endometriosis\n. Eur J Obstet Gynecol Reprod Biol. 2001,\n96:21-34. \n10.1016/s0301-2115(00)00405-x\n10\n. \nBagan P, Le Pimpec Barthes F, Assouad J, Souilamas R, Riquet M: \nCatamenial pneumothorax: retrospective\nstudy of surgical treatment\n. Ann Thorac Surg. 2003, 75:378-81; discusssion 381. \n10.1016/s0003-\n4975(02)04320-5\n11\n. \nChen Y, Zhu HL, Tang ZW, et al.: \nEvaluation of circulating endometrial cells as a biomarker for\nendometriosis\n. Chin Med J (Engl). 2017, 130:2339-45. \n10.4103/0366-6999.215325\n12\n. \nMok-Lin EY, Wolfberg A, Hollinquist H, Laufer MR: \nEndometriosis in a patient with Mayer-Rokitansky-\nKüster-Hauser syndrome and complete uterine agenesis: evidence to support the theory of coelomic\nmetaplasia\n. J Pediatr Adolesc Gynecol. 2010, 23:e35-7. \n10.1016/j.jpag.2009.02.010\n \n2024 Ávila et al. Cureus 16(10): e71202. DOI 10.7759/cureus.71202\n6\n of \n6","source_license":"CC0","license_restricted":false}