{"paper_id":"a3ef0f07-4bef-4418-88c8-872ca1d75dc2","body_text":"www.jpccr.euCASE STUDY\nPulmonary complications requiring surgical \nintervention caused by endometriosis  – case \nstudies\nKamila Stopińska1,A-D \n  , Mariusz Kazimierz Wójtowicz1,E-F \n , Karolina Marczak1,B,D \n ,  \nOlga Grzelak1,B,D \n1 Women’s and Child Health Centre, Zabrze, Poland  \nA – Research concept and design, B – Collection and/or assembly of data, C – Data analysis and interpretation,  \nD – Writing the article, E – Critical revision of the article, F – Final approval of the article\nStopińska K, Wójtowicz MK, Marczak K, Grzelak O. Pulmonary complications requiring surgical intervention caused by endometriosis – case \nstudies. J Pre-Clin Clin Res. 2022; 16(4): 145–148. doi: 10.26444/jpccr/157432\nAbstract\nEndometriosis is a condition described as growth of the endometrium outside the uterine cavity. Lesions can occur in many \nareas of the body, including the pleural cavity and lungs. Etiology of this condition is still unknown. Two medical cases are \ndescribed: a 47-year-old patient reporting chest pain, who had resection of the apex of the right lung a year earlier,and \n41-year-old patient with frequent lower abdominal pain and recurrent haemoptysis for about 16 years, which had been the \ncause of multiple hospitalizations in pulmonary departments. Both patients underwent several examinations – tomography, \nMRI, and surgical diagnostics. In both cases, the tests showed the presence of thoracic endometriosis. The described cases \nindicate diagnostic and therapeutic difficulties in patients with thoracic endometriosis. In conclusion, treatment of the \nmanifestation of thoracic endometriosis is a great challenge because of limitation of the possibilities for surgical procedures \nand non-invasive diagnostic tests.\nKey words\nendometriosis, thoracic endometriosis, catamenial pneumothorax, catamenial haemoptysis\nAbbreviations\nCT – computed tomography; VATS – video-assisted thoracoscopic surgery; GnRH – gonadotropin-releasing hormone; \nTES – thoracic endometriosis syndrome; TVUS – transvaginal ultrasound\nINTRODUCTION\nEndometriosis is described as growth of the endometrium \noutside the uterine cavity and is associated with characteristic \nsymptoms, such as dysmenorrhea, menorrhagia, and chronic \npelvic pain, and causes infertility [1]. The disease affects \n15% among reproductive-age women [2, 3]. Endometriosis \nhas a strong impact on psychological and social well-being, \ncausing depression, sexual dysfunction and inability to work. \nTreatment also generates significant costs (long diagnosis, \nsurgical therapy, hospital admission, fertility therapy) [4].\nLaparoscopy is the gold standard in the diagnostic process. \nOne of the main reasons for the delay of a final diagnosis of \nthis condition is because it requires surgery and histological \nexamination of the tissue [4,5]. Endometric lesions may present \nin many places in the pelvis, including the uterus (adenomyosis), \novary (endometrioma), pelvic peritoneum, bladder/ureter, \nrectum, colon, uterosacral ligaments, rectovaginal septum, \nvaginal wall, or pouch of Douglas. Endometrial implants may \nalso occur at many rare locations in the body, such as the lungs, \nliver, pancreas, brain and C-section scar, resulting in a variety \nof symptoms related to these organs [4]. The most common \nlocation of endometriosis outside the abdominopelvic cavity \nis the thoracic cavity [6]. In 80% of cases, lesions are located \non the right side [7, 8].\nThe presence of functional endometrial tissue in the thoracic \ncavity is called thoracic endometriosis syndrome (TES), an \nextremely rare disease with complex causes and pathogenesis \nthat remains unclear [7, 9], and can be classified as pulmonary \nor pleural [8]. The pathogenesis of this disease is still unknown \nalthough several concepts have been presented, but none of \nthem can fully explain all the clinical manifestations of the \nsyndrome [9]. Retrograde menstruation is the most prominent \ntheory, which assumes that endometrial cells  undergo a \nretrograde movement through the fallopian tubes into the \nperitoneal cavity, and implant on peritoneal surfaces [6, 10]. \nThe coelomic metaplasia theory assumes that endometriosis \nis formed by metaplasia of mesothelial cells lining the pleura \nand peritoneal surfaces into endometrial glands and stroma. \nTransformation of these cells may be stimulated by estrogens \n[6]. Although the coelomic metaplasia theory may provide \nan explanation for  pleural cases of endometriosis, the \nbronchopulmonary lesions remain unexplained [6, 8].\nOn the other hand, a possible explanation for broncho -\npulmonary endometrial lesions is the  theory of benign \nmetastases, which proposes that ectopic endometrial implants \nare the result of lymphatic or haematogenous dissemination \nof endometrial cells [2, 6]. The final approach  explaining \nTES involves prostaglandin F2α which is detectable in the \nplasma of women during menstruation. Prostaglandin F2α \nis a constrictor of bronchioles and blood vessels, which \nincreases during menstruation and may lead to alveolar \nrupture of previously formed subpleural blebs and bullae, \nresulting in a pattern of catamenial pneumothorax [7, 10].\n Address for correspondence: Kamila Stopińska, Women’s and Child Health \nCentre, Zabrze, Poland\nE-mail: kamilastopinska1995@gmail.com\nReceived: 11.10.2022; accepted: 13.12.2022; first published: 27.12.2022\nJournal of Pre-Clinical and Clinical Research 2022, Vol 16, No 4, 145-148\n\nKamila Stopińska, Mariusz Kazimierz Wójtowicz, Karolina Marczak, Olga Grzelak . Pulmonary complications requiring surgical intervention caused by endometriosis …\nPelvic endometriosis expressions typically occur \napproximately 5–7 years before developing manifestations \nof thoracic endometriosis [6].  Symptoms of pulmonary \nendometriosis are always associated with the onset of menses \nand usually manifests as catamenial haemoptysis  [8,11]. \nHaemoptysis in the majority of cases stop after the cessation \nof menstruation and may be accompanied by chronic cough, \ncatamenial pneumotxorax, episodes of low grade fever which \nmay be recurrent, and asymptomatic lung nodules [3].\nOBJECTIVE\nThe presented case reports aim to demonstrate the \ndiagnostic and therapeutic difficulties in patients with \nthoracic endometriosis, and also to indicate the difficulties \nin conducting therapy and the need for a multidisciplinary \nand individual approach to the patient.\nMATERIALS AND METHOD\nThe material for this case study was collected from the real-\nlife clinical process and medical records of the gynaecological \nward at the Women’s and Child Health Centre in Zabrze, \nPoland.\nIn accordance with the Helsinki Declaration, the case \nreports were fully anonymised, and none of the data presented \nwould make identification of the patients possible. Under \nPolish law, such case reporting does not require the consent \nof a Bioethics Committee.\nCASE STUDIES\nPatient 1\nA 47-year-old patient treated chronically for endometriosis \nreported to the attending physician complaining of pain in \nthe right side of the chest. Due to the ailments described by \nthe patient, a chest X-ray and a CT chest scan were performed. \nIn the right pleural cavity, fluid,  atelectasis and fibrous \nchanges were visualized. At the time of reporting to the \nattending physician, the patient complained of a stinging pain \nin the chest while breathing and performing  movements, \nand reduced exercise tolerance. The attending physician \nordered a pelvic MR examination which showed the features \nof intensified deeply infiltrating endometriosis, with ingrown \nlesions in the dorsal part of the sigmoid colon and the area \nof the large intestine at the border of the sigmoid colon and \nrectum. Surgical consultation was recommended.\nThe  patient was referred to the thoracic surgery ward \nwhere right-sided  recurrent pneumothorax with exudate \nto the pleural cavity was diagnosed. The pneumothorax \nwas  decompressed by drainage of the pleural cavity. Past \nmedical history included resection of  the apex of the \nright lung a year earlier. The material collected during \nthe operation  confirmed endometriosis of the lungs and \npleura. The attending physician initiated treatment with the \nDiphereline SR (Triptorelinum) in a dose of 3.75 mg with \nprolonged release administered intramuscularly. After the \nadministration of six doses of Diphereline, treatment with \nDepo-Provera (medroxyprogesteroni acetas) was initiated \nby intramuscular injection every 90 days.\nAt the follow-up visit six months later, the patient did \nnot report any pain in the pelvic area, nor the symptoms \nof pneumothorax described earlier. During the visit,  a \ntransvaginal pelvic ultrasound was performed which showed \na tumour located behind the uterus that may represent an \nendometriosis tumour (Fig. 1). It was therefore decided to \ncontinue the supply of Depo-Provera. The treatment cycle \nwas completed after the eighth intramuscular injection of \nDepo-Provera. The last medical appointment took place in \n2021. The patient was in good general condition and did not \nreport any complaints.\nFigure 1. Patient 1.Transvaginal pelvic ultrasound, uterus, a tumour located behind \nthe uterus that may represent an endometriosis tumour.\nPatient 2\nThe 41-year-old patient had been hospitalized several times \ndue to pain in the lower abdomen, and reported suffering \nperiodic haemoptysis for about 16 years, which resulted \nin repeated hospitalization in pulmonary departments. \nEndometriosis was suspected and confirmed by laparoscopy \nin 2013. The patient described the pain in the lower abdomen \nas jerking, stinging, pulling, burning, rushing, but not related \nto the phase of the menstrual cycle. Pain and haemoptysis \nmade it difficult to perform daily activities. The patient \nunderwent multiple surgical consultations due to repeated \nepisodes of severe lower abdominal pain with suspected \nacute abdomen.\nTransvaginal pelvic ultrasound showed an adhesion and \nintrauterine endometrial lesions (Fig. 2, Fig. 3). Repeated \nhaemoptysis was the basis for extending pulmonary \ndiagnostics. In 2018, the decision was made to re-laparoscopy \nFigure 2. Patient 2. Transvaginal pelvic ultrasound, ovary, adhesion\n146 Journal of Pre-Clinical and Clinical Research 2022, Vol 16, No 4\n\nKamila Stopińska, Mariusz Kazimierz Wójtowicz, Karolina Marczak, Olga Grzelak . Pulmonary complications requiring surgical intervention caused by endometriosis …\nto release the adhesions, and perform electrocautery of the \nright ovary. Endometriosis foci in the small pelvis were \ncoagulated. A CT of the chest was ordered which showed a \nlesion that could correspond to the endometriosis lesion in \nsegment VI. Due to the suspicion of thoracic endometriosis, it \nwas decided to perform interventional bronchoscopy. Due to \npersistent pain in the lower abdomen, haemoptysis and heavy \nbreathing, treatment with Diphereline SR (Triptorelinum) \nin a dose of 3.75 mg was started for six months\nDISCUSSION\nThe presented cases describe the diagnostic and therapeutic \ndifficulties in patients with pulmonary endometriosis. The \nresults were compared with currently available literature in \nwhich it was noticed that in the described cases difficulties \nwere also experienced in diagnosing the disease. Bricelj et al. \nconducted a systematic review and reported that 39.5% of \npatients with endometrial pneumothorax had coexisting \npelvic endometriosis [12]. The same study reported that people \ndiagnosed with pelvic endometriosis had more endometrial \nimplants in the chest [12]. Despite the implementation of \nappropriate treatment and surgery, symptoms recurred \nin 26.9% of patients, [12]. Kardaman et al., in case report \ndescribe the clinical case of a 48-year-old female patient \nwith recurrent pneumothorax on the right side. The authors \ndescribe that endometrial changes were more common on the \nright side, which was also found in Patient 1 [13]. Researchers \nfrom the Pneumology Department, Farhat Hached Hospital \nin Sousse, Tunisia, described the case of a 42-year-old \nwoman with recurrent right-sided pneumothorax. She \nunderwent video-assisted thoracoscopic surgery (VATS), \nduring which numerous endometrial lesions were visualized \nand confirmed by histopathological examination [11]. The \npatient was treated with a GnRH agonist, as was the case \nwith Patient 1. Also in this case, no permanent remission \nwas achieved, and the symptoms recurred after 22 months \n[11]. The case report by Leonardo-Pinto et al. also describes \npulmonary manifestations of endometriosis [14] in the case \nof a 23-year-old female patient with haemoptysis occurring \nfor two years around the time of menstruation. A nodule was \nfound in the right lung that might correspond to a lesion of \nendometriosis; transvaginal ultrasound confirmed deeply \ninfiltrating endometriosis of the reproductive organs. Surgical \ntreatment was not performed in this patient. Based on the \nsymptoms, empirical treatment was implemented, achieving \nan improvement in the clinical condition [14]. Researchers \nfrom the Department of Thoracic and Cardiovascular \nSurgery, Uijeongbu St. Mary’s Hospital, The Catholic \nUniversity of Korea in Seoul, Republic of Korea, describe \nsurgical intervention as an effective means of preventing \nthe recurrence of haemoptysis, based on the case report \nof a 23-year-old patient with catamenial haemoptysis [15]. \nAfter resection of the diseased lung fragment, no recurrence \nof clinical manifestations of the disease was observed \nduring the 5-year follow-up. [15]. The current case reports \nconfirms the systematic review reported by researchers from \nthe Department of Obstetrics and Gynecology at McGill \nUniversity in Montreal, Canada. Observations in the current \ncases concurr – the most common pulmonary endometriosis \noccurs on the right side, and are also in agreement with the \nliterature [16].\nLimitations of the case studies. In the two case reports it may \nbe that the patients had different lengths of medical history, as \nwell being of different ages. Currently, two more patients with \ncatamenial haemoptysis are being monitored at the Women’s \nand Child Health Center in Zabrze; therefore, expansion \nis planned in which further studies will be undertaken to \ncheck whether the occurrence of pulmonary complications in \npatients with endometriosis can be predicted before clinical \nmanifestations occur.\nFunding. The article was not funded by any external sources\nDisclousures. The authors declare that there are no conflicts \nof interest to disclose\nFigure 3. Patient 2.  Transvaginal pelvic ultrasound, uterus, intrauterine endo -\nmetrium\nTable 1. \nCase Timeline Event\nPatient 1 1998 Surgical resection of ovarian lesions.\n 2019 VATS surgery - resection of the apex of the right lung, \npleurectomy and pleurodesis.\n 2019 Lesions in TVUS – suspicion of endometrial tumour.\n 2020 Pneumothorax - histopathological examination confirmed \nthoracic endometriosis.\n 2022 Lesions of pleura in CT – suspicion of pleural endometriosis.\n 2022 Pneumothorax - hospitalization in the thoracic surgery \ndepartment, decompression and drainage.\n 2022 TVUS – normal results.\nPatient 2 2004 First symptoms: lower abdominal pain.\n 2013 First laparoscopy due to endometriosis.\n 2014 Endometrial cyst of the left ovary in TVUS.\n 2017–\n2019\nMultiple hospitalization for lower abdominal pain.\n 2018 Second laparoscopy due to endometriosis.\n 2022 Bronchial nodule in CT – suspicion of thoracic \nendometriosis. \n 2022 Interventional bronchoscopy.\n147Journal of Pre-Clinical and Clinical Research 2022, Vol 16, No 4\n\nKamila Stopińska, Mariusz Kazimierz Wójtowicz, Karolina Marczak, Olga Grzelak . 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