{"paper_id":"bd0d083d-5046-461b-8870-e65713b41126","body_text":"Takayama et al. \nGeneral Thoracic and Cardiovascular Surgery Cases            (2023) 2:90  \nhttps://doi.org/10.1186/s44215-023-00110-w\nCASE REPORT Open Access\n© The Author(s) 2023. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which \npermits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the \noriginal author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or \nother third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line \nto the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory \nregulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this \nlicence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecom-\nmons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.\nGeneral Thoracic and\nCardiovascular Surgery Cases\nMeigs syndrome after treatment \nfor catamenial pneumothorax\nMao Takayama1*  , Tetsuya So1, Naoki Yamashita1, Masatoshi Yamaoka2, Takashi Yoshimatsu2 and \nTsuneiro Oyama3 \nAbstract \nIn the field of thoracic surgery, catamenial pneumothorax (CP) is known as a disease peculiar to women, but it \nis rare among female pneumothoraces and is rarely encountered in clinical practice. Meigs syndrome (MS) \nis another female-specific disease, defined as a benign ovarian tumor with pleural and ascites effusions, but it is rare \nand the details of the pathogenesis of MS have not yet been elucidated.\nA 40-year-old Japanese woman came in with dyspnea. Chest radiography revealed a collapsed right lung. She \nunderwent video-assisted thoracoscopic surgery, which revealed multiple diaphragmatic foramens. She was therefore \ndiagnosed with CP . Later, when she was 50-year-old, returned with chest pain. Computed tomography of the chest \nand abdomen showed right massive pleural effusion and a large tumor and ascites in the pelvis. This condition \nwas suggestive of MS. The patient underwent bilateral oophorectomy, and the right pleural effusion and ascites \nresolved promptly after surgery.\nIn conclusion, both menstrual-associated CP and MS are very rare conditions, and to the best of our knowledge, there \nare no reported cases of the combination of the two. However, it is possible that some MS patients may have MS \nwithout CP , and the present case is considered to be valuable because a small diaphragmatic foramen was identified \nvia thoracoscopy, which has been a minority opinion among the mechanisms of pleural effusion in MS.\nKeywords Meigs syndrome, Catamenial pneumothorax, Ovarian tumors\nIntroduction\nCatamenial pneumothorax (CP) is known in the field of \nthoracic surgery as a disease peculiar to women. CP is \nthe most common manifestation of thoracic endome -\ntriosis. Because CP is a rare disease, it is rarely encoun -\ntered in clinical practice. Conversely, Meigs syndrome \n(MS) is also a disease specific to women and is defined \nas a benign ovarian tumor with pleural effusion and \nascites. MS is also a rare disease, and the details of the \nmechanism underlying MS have not yet been elucidated. \nHerein, we report an extremely rare case of CP followed \nby MS.\nCase report\nA 40-year-old Japanese woman presented to our hos -\npital (Shinkomonji Hospital, Kitakyushu, Japan) due \nto dyspnea and right-sided chest pain. These symp -\ntoms occurred within 48 h after the onset of menstrua -\ntion. Chest radiography revealed a collapsed right lung \n(Fig.  1). Chest computed tomography (CT) showed no \npleural effusion and no emphysematous changes or neo -\nplastic lesions (data not shown); thus, CP was suggested. \n*Correspondence:\nMao Takayama\n0m0xolo@gmail.com\n1 Department of Thoracic Surgery, Shinkomonji Hospital, 2-5 \nDairishinmachi, Moji-Ku, Kitakyushu 800-0057, Japan\n2 Department of Thoracic Surgery, Fukuoka-Wajiro Hospital, 2-2-75 \nWajirogaoka, Higashi-Ku, Fukuoka 811-0213, Japan\n3 Department of Surgery, Imamitsu Homecare Clinic, 1-9-10 Imamitsu, \nWakamatsu-Ku, Kitakyushu 808-0071, Japan\n\nPage 2 of 5Takayama et al. General Thoracic and Cardiovascular Surgery Cases            (2023) 2:90 \nAlthough we performed right thoracic drainage, minor \nair leakage continued. The patient underwent video-\nassisted thoracoscopic surgery. Thoracoscopy revealed \nmultiple diaphragmatic foramens around the central \ntendon (Fig.  2A); however, no abnormal legions were \nnoted at the visceral pleura or wall-side pleura. Intraop -\nerative water sealing test showed no obvious leak and no \npartial excision was performed. It is difficult to perform \ncomplete thoracoscopic resection of the diaphragm, and \nhormone therapy is fundamental to this disease, and \nsurgery is not performed in consideration of the degree \nof invasiveness. The entire surface of the diaphragm was \ncovered with a polyglycolic acid (PGA) sheet (Fig.  2B) \nand fibrin glue.\nAs a result of consultation with an obstetrician and \ngynecologist using the clinical course and intraopera -\ntive findings, the patient was thus diagnosed with CP \nand underwent hormonal therapy with danazol. No \nclinical recurrence of CP was noted thereafter.\nTen years later (at the age of 50 years), the patient \nreturned to our hospital with a complaint of chest pain. \nChest radiography revealed a right massive pleural effu -\nsion (Fig.  3). Chest CT showed pleural effusion and pas -\nsive atelectasis (Fig.  4A). Abdominal CT detected ascites \n(Fig. 4B) and a huge tumor with a regular margin meas -\nuring 10 × 9 cm in size at her pelvis (Fig.  4C). Abdominal \nmagnetic resonance imaging (MRI) revealed hypointense \nsignals on T1-weighted MRI (Fig.  4D) and hyperintense \nsignals on T2-weighted MRI (Fig.  4E). The carbohydrate \nantigen 125 level was 645 U/mL (reference range: 0–35 \nU/mL). The combination of symptoms, including right \npleural effusion, ascites, and ovarian tumor, led to the \nFig. 1 Chest radiography showing right pneumothorax\nFig. 2 Surgical findings. A Multiple diaphragmatic foramens. B A PGA sheet was applied to the entire diaphragm\nFig. 3 Chest radiography revealed a right massive pleural effusion\n\nPage 3 of 5\nTakayama et al. General Thoracic and Cardiovascular Surgery Cases            (2023) 2:90 \n \nsuspicion of MS. Therefore, we referred the patient to \nthe Department of Gynecology, where gynecologists per -\nformed bilateral tubal oophorectomy. Histopathological \nexamination of the resected tumors confirmed ovarian \nfibroma and no evidence of malignancy. After gynecologic \nsurgery, a rapid disappearance of right pleural effusion and \nascites was noted. The clinical course also suggested MS.\nDiscussion\nCP was first reported by Maurer in 1958 and remains a \nrelatively rare disease [1]. The diagnostic criteria for CP \nis its appearance before or within 72 h after the start of \nmonthly bleeding. The broad definition of CP is a pneu -\nmothorax occurrence from 7 days before the start of \nmonthly bleeding to 7 days after the end of bleeding. \nAdditional criteria include characteristic pleural lesions, \nright-sided location of the pneumothorax, and concomi -\ntant endometriosis.\nResearchers have proposed three theories for the \npathogenesis of CP . First, Mauer proposed a mechanism \nin which endometrial tissue is seeded into the thoracic \ncavity via a small foramen in the diaphragm and grows on \nthe visceral pleura, resulting in pneumothorax with men -\nstruation [2]. He also suggested that after endometrial \nFig. 4 CT. A Chest: pleural effusion, passive atelectasis. B, C Abdomen and pelvis: ascites and a huge tumor 10 cm in diameter in the pelvis. MRI. D \nHypointense signals on T1-weighted MRI. E Hyperintense signals on T2-weighted MRI\n\nPage 4 of 5Takayama et al. General Thoracic and Cardiovascular Surgery Cases            (2023) 2:90 \ntissue is deposited on the diaphragm, its shedding dur -\ning menstruation forms a small foramen, resulting in \npneumothorax. Second, Lillington reported that endo -\nmetrial tissue migrates hematogenously into the periph -\neral airways, forming a bulla or bleb by the check valve \nmechanism, resulting in pneumothorax [3]. Third, Rossi \nsuggested that high levels of circulating prostaglan -\ndin F2a, resulting from a collapsed endometrium, cause \nvasoconstriction and bronchospasm, leading to the rup -\nture of the alveolar tissue [4].\nMauer’s theory is supported by the identification of \nendometriosis in the diaphragm in most patients with \npneumothorax. However, these theories alone can -\nnot explain all cases of CP without foramens in the dia -\nphragm. Such cases may be caused by the mechanism \ndue to Lillington and/or Rossi’s theory. In this case, small \nholes in the diaphragm were observed by thoracoscopic \nsurgery 10 years previously.\nMS was first reported by Meigs in 1937, who examined \na series of seven patients with ascites and pleural effusion \nassociated with benign ovarian fibroma [5]. Subsequently, \nthis condition was defined as MS, with the following four \ncharacteristics: (1) benign ovarian fibroma or fibroma-\nlike tumor, (2) ascites, (3) pleural effusion, and (4) rapid \nresolution of ascites and pleural effusion after tumor \nremoval [6]. The pathogenesis of ascites and pleural effu -\nsion in MS remains unknown. Hamilton described that \nascites may result from edematous fibromas that leak \nfluid or the increased lymphangial pressure in the abdo -\nmen and pelvis caused by the tumor itself. The lymphat -\nics are abundant on the right side of the diaphragm, \nand pleural effusion is often observed on the right side \n[7]. Conversely, Okuda suggested that pleural effusion \narises when ascites moves from the peritoneal cavity to \nthe pleural cavity through diaphragmatic defects [8]. The \nlatter theory is supported by the valuable case presented \nherein, in which small holes in the diaphragm were iden -\ntified during the CP operation 10 years previously.\nDespite robust hormonal therapy for CP , the patient \ndeveloped MS. In this case, the diaphragm was fully cov -\nered with a PGA sheet for small holes in the diaphragm \nat the time of the CP operation. According to the instruc-\ntion manual, a PGA sheet is almost completely absorbed \nin approximately 15 weeks. At the time of the CP opera -\ntion, we believed that CP was a systemic gynecologic \ndisease that was treated mainly via hormonal therapy. \nFurthermore, total resection and reconstruction of the \ndiaphragm were highly invasive. Therefore, we decided \nto place the PGA sheet on the diaphragm for coverage. \nMurakami reported a case of artificial pericardial rein -\nforcement using an expanded polytetrafluoroethylene \n(ePTFE) sheet for diaphragmatic hernia after CP dia -\nphragmatic excision [9]. Because the CP diaphragm is \nvery fragile, ePTFE sheets are considered to provide \nsecure closure with sufficient strength. Presently, the \nmost suitable surgical procedure for CP appears to be \nthe total replacement of the diaphragm using the ePTFE \nsheet.\nIn this way, this patient developed MS unexpectedly, \nand the present case confirms the hypothesis of right \npleural effusion in MS. Because endometriosis is a com -\nmon gynecologic disease and is said to be increasing in \nrecent years, it is possible that CP in a patient with endo -\nmetriosis was mixed in with MS, in which no endome -\ntriosis was noted. Through this study, we believe that \nregular gynecological examinations are necessary for CP \npatients.\nIn conclusion, both menstrual-associated CP and MS \nare very rare conditions, and to the best of our knowl -\nedge, there are no reported cases of the combination of \nthe two. The present case is considered to be valuable \nbecause a small diaphragmatic foramen was identified via \nthoracoscopy, which has been a minority opinion among \nthe mechanisms of pleural effusion in MS.\nAbbreviations\nCP  Catamenial pneumothorax\nCT  Computed tomography\nMS  Meigs syndrome\nPGA  Polyglycolic acid\nMRI  Magnetic resonance imaging\nePTFE  Expanded polytetrafluoroethylene\nAcknowledgements\nWe have no person who supported our manuscript.\nAuthors’ contributions\nNot applicable.\nFunding\nNot applicable.\nAvailability of data and materials\nNot applicable.\nDeclarations\nEthics approval and consent to participate\nNot applicable.\nConsent for publication\nNot applicable.\nCompeting interests\nThe authors declare no competing interests.\nReceived: 22 December 2022   Accepted: 29 July 2023\nReferences\n 1. Marjański T. Catamenial pneumothorax – a review of the literature. Thorac \nSurg. 2016;13(2):117–21.\n\nPage 5 of 5\nTakayama et al. General Thoracic and Cardiovascular Surgery Cases            (2023) 2:90 \n \n•\n \nfast, convenient online submission\n •\n  \nthorough peer review by experienced researchers in your field\n• \n \nrapid publication on acceptance\n• \n \nsupport for research data, including large and complex data types\n•\n  \ngold Open Access which fosters wider collaboration and increased citations \n \nmaximum visibility for your research: over 100M website views per year •\n  At BMC, research is always in progress.\nLearn more biomedcentral.com/submissions\nReady to submit y our researc hReady to submit y our researc h  ?  Choose BMC and benefit fr om: ?  Choose BMC and benefit fr om: \n 2. Maurer ER, Schaal JA, Mendez FL. Chronic recurring spontaneous \npneumothorax due to endometriosis of the diaphragm. J Am Med Assoc. \n1958;168:2013–4.\n 3. Lillington GA, Mitchell SP , Wood GA. Catamenial pneumothorax. JAMA. \n1972;219:1328–32.\n 4. Rossi NP , Goplerud CP . Recurrent catamenial pneumothorax. Arch Surg. \n1974;109:173–6.\n 5. Corsellis JAN, Goldberg GJ, Norton AR. “Limbic encephalitis” and its asso-\nciation with carcinoma. Brain. 1968;91:481–96.\n 6. Meigs JV, Cass JW. Fibroma of the ovary with ascites and hydrothorax \nwith a report seven cases. Am J Obstet Gynecol. 1937;33:249–67.\n 7. Okuda K. A case of Meigs’ syndrome, with special reference to the mecha-\nnism of pleural effusion. Int Med. 1967;20:569.\n 8. Meigs JV. Fibroma of the ovary with ascites and hydrothorax Meigs’ \nsyndrome. Am J Obstet Gynecol. 1954;67:962–85.\n 9. Murakami T, et al. Post-operative diaphragmatic hernia in a patient with \ncatamenial pneumothrax. J Clin Surg. 2011;66(5):701–4.\nPublisher’s Note\nSpringer Nature remains neutral with regard to jurisdictional claims in pub-\nlished maps and institutional affiliations.","source_license":"CC0","license_restricted":false}