A Case of Death of Patient With Ovarian Fibroma Combined With Meigs Syndrome and Literature Review

A Case of Death of Patient With Ovarian Fibroma Combined With Meigs Syndrome and Literature Review | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case Report A Case of Death of Patient With Ovarian Fibroma Combined With Meigs Syndrome and Literature Review Na Tan, Kai-yuan Jin, Xiao-rong Yang, Cheng-fang Li, Jin Yao, and 1 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-1374873/v1 This work is licensed under a CC BY 4.0 License Status: Under Review Version 1 posted 7 You are reading this latest preprint version Abstract Ovarian fibroma is the most common benign pure stromal tumor. It has no specific clinical manifestation, most of which are pelvic or adnexal masses. 10%-15% of cases with hydrothorax or ascites, after tumor resection, hydrothorax and ascites disappear, known as Meigs Syndrome. The elevated level of CA125 in a few patients was easily misdiagnosed as ovarian malignant tumor. A case of bilateral Ovarian fibroma associated with Meigs Syndrome is reported and the literature is reviewed in order to improve the understanding of the changes and avoid misdiagnosis. ovarian fibroma Meigs Syndrome Figures Figure 1 Figure 2 Figure 3 Introduction A 46-year-old female was presented with recurrent abdominal distension and pain for 4 years. The symptoms worsened with vomiting for 1 month, and the weight loss was 5 kg recently. The diagnosis before admission: duodenal stenosis, pyloric obstruction? ovarian cancer? Observations by ultrasound: bilateral adnexal mass, pelvic and peritoneal effusion. Plain and enhanced CT scan of the abdomen (see Fig. 1): there were multiple irregular cystic and solid masses in bilateral adnexa, considered neoplastic lesions. It should think about the possibility of ovarian cancer. Moreover, a large amount of effusion was found in the bilateral thoracic cavity and pelvic and abdominal cavity. There was effusion in abdominal soft tissue. Female tumor-associated antigen = CA19-9 41.10 U/mL and CA125 56.5 U/mL (normally < 35 U/mL). Biochemical tests of ascites: total proteins 42.3 g/L, rivalta test (+). The clinical diagnosis: causes of abdominal distension: metastasis and invasion of ovarian cancer? pyloric obstruction? multiple abdominal metastases? Intraoperative observation: there exited adhesion with a hard texture between the middle segment of the greater omentum and lower margin of the right hepatic lobe and between the duodenal bulb and pyloric part. A large amount of pale blood fluid could be found under the left subphrenic space, right liver lobe and pelvic cavity. The bilateral ovaries showed nodular and beaded changes, with multiple nodules. The nodules had different sizes, with slightly hard quality and acceptable mobility. Intraoperative frozen sections: spindle cell tumor. It was considered the diagnosis of sex cord-stromal tumor. Subtotal gastrectomy and gastrojejunostomy + distal duodenal exclusion + complex enterolysis + bilateral accessory and hysterectomy were performed. The patient frequently vomited after the operation, and 1500 mL liquid was drained from the gastric tube. The patient had a fever and a CT scan of the chest showed increased pleural effusion. There were secondary coagulation dysfunction, sepsis and multiple organ failure. Pathological Examination Observation by the naked eye: the sizes of bilateral ovaries were 8×6×4 cm 3 and 7×5×2 cm 3 , respectively. The surface of the ovaries was smooth with multinodular and beaded changes. The size of the nodules varied, with the largest diameter of 3 cm (see Fig. 2 ). The surface section was off-white, with moderate hardness and solid structure. The ovaries were cystic locally and the thickness of the cyst wall was 0.1 cm with colloidal substances inside. There was no bleeding or necrosis. Microscopic examination (see Fig. 3 ): the ovarian structure disappeared. A large number of spindle cells or oval cells are arranged in feathery, braided, or whirl-like, occasionally storiform. The nuclei of tumor cells were long fusiform without mitotic figures. There was no lipid in the cytoplasm. The intercellular edema was prominent without mucoid degeneration, hyaline degeneration, calcification and ossification. The results of immunohistochemistry showed that tumor cells: Vimentin, WT-1, Calretinin (partial expression), Ki-67 (3% positive rate), α-inhibin (+), reticular fiber dyeing (+). Pathological examination: ovarian fibroma. Discussion According to the definition of female reproductive system diseases proposed by WHO in 2020, ovarian fibroma is a benign pure stromal tumor composed of the spindle to oval fibroblasts like cells that produce collagen. Ovarian fibroma is the most common sex cord-stromal tumor, accounting for 4% of all ovarian tumors. [ 1 ] It can occur at any age, but it is most common in middle age (average 48 years old). It seldom occurs before 30 years old and has also been reported in infants of 7 months old. [ 2 ] Most of the tumors are unilateral and rarely bilateral. The tumor’s average size is 8 cm, about 1/3 of the tumors can be less than 3 cm. There was no specific clinical manifestation and most of them show pelvic or adnexal masses. [ 3 ] About 10%-15% of ovarian fibroma can be combined with pleural fluids or ascites, described as Meigs syndrome. It was reported by Meigs and Cassa in 1937. In 1954, Meigs Syndrome was defined as a benign ovarian fibroma or fibroid tumor with pleural fluids and ascites. The pleural fluids and ascites disappeared after tumor resection, and ascites recurred in tumors with a diameter > 10 cm. The possible cause of pleural fluids and ascites is that the fluid within tumors infiltrates the abdominal cavity through the serosal surface. It then enters into the unilateral or bilateral pleural cavity through the lymphatic vessels or the communication between the pleural cavity and the abdominal cavity. [ 4 ] There are no specific markers for accurate diagnosis of ovarian fibroids/fibroids before surgery. [ 5 ] CA125 level in serum increased in a few patients. Some studies suggested that increased CA125 level in serum might be related to some biochemical factors, such as elevated intraperitoneal pressure caused by mechanical stimulation of ascites to the peritoneum, or peritoneal mesothelial cells, which might be misdiagnosed as endometriosis or malignant ovarian tumor. [ 6 ] In this case, the patient had an elevated CA125 and ascites before the operation, which was easily misdiagnosed as a malignant ovarian tumor. According to the proportion of fibroblasts and collagen fibers, ovarian fibroma can be divided into cellular and fibrous types. Cellular fibroma is associated with ovarian rupture and extraovarian adhesions and has a risk of local recurrence. Besides, fibroma has some particular subtypes. Fibroma with a small amount of sex cord components refers to less than 10% tubules with different differentiation degrees or incomplete tubular structure of immature supporting cells in fibroma. Moreover, there are abundant tumor cells arranged closely with unclear cytoplasmic boundaries and fewer collagen components. The nuclear chromatin increases and is in round or oval with or without mild atypia, and mitotic rates are ≥ 1-3/10 HPFs. In this case, it can be called cell-rich fibroma. Cell-rich fibroma commonly presents a benign clinical course but has low malignant potential and occasional local recurrence. [ 7 , 8 , 9 ] It should be differentiated from the following diseases: Thecoma: thecoma is another kind of pure stromal tumor. However, fibroma and thecaoma have overlapping morphological features. Some cases that are difficult to distinguish can be called fibrothecoma or follicular fibroma. Microscopically, several relatively isolated theca cells rich in lipid between fat spindle-shaped fibroblasts or fibroblasts, showing clump distribution. However, if the patient has no steroid hormone changes and the composition of theca cells in tumors is less than 10% with negative immunohistochemical inhibin, it is not recommended to be diagnosed as fibrothecoma but directly diagnosed as a fibroma. Theca cells in this patient are not rich in lipid and have loose fibrous stroma with prominent edema. Massive edema of the ovary: when fibroma shows obvious edema, it should be differentiated from this disease. Most of the patients with massive edema of the ovary are younger than 30 years old, and abdominal pain is the main clinical symptom. Ovaries have the normal structure, and the whole ovary is diffuse with edema, and sometimes luteinized cells can be found in the stroma. The patient, in this case, does not have ovaries with the normal structure. Fibrosarcoma: the tumor cells with moderate to severe atypia are very abundant, arranged in braided shape, with eosinophilic cytoplasm and unclear boundaries. The nuclei are in round or fusiform shape, with abundant chromatin, prominent nucleolus. Mitotic images are common, with an average of > 4/10 HPFs. There usually are bleeding and necrosis complications. The transitional zone between fibroma and fibrosarcoma can be found in a few tumors. Fibrosarcoma has a poor prognosis and a few patients had been reported to survive more than 5 years. The identification of Meigs Syndrome: Pseudo Meigs Syndrome: in addition to ovarian fibroma, ascites and pleural fluids occur in patients with other benign or malignant tumors in the pelvis or abdomen. [ 10 ] Pseudo-Pseudo Meigs Syndrome: it is also known as Tajalma Syndrome. It can be found in patients with systemic lupus erythematosus accompanied by ascites, pleural fluids, and elevated CA125 level in serum. [ 11 ] The patient had ovarian fibroma with pleural fluids and ascites, accompanied by slightly elevated CA125. There were no other tumors or systemic lupus erythematosus. After bilateral accessory and hysterectomy, the pleural fluids and ascites disappeared temporarily, consistent with Meigs Syndrome. The subsequent recurrence of pleural fluids and ascites was due to severe infection rather than the disease itself. Based on the ultrasound imaging, the typical ovarian fibroma can be manifested as a well-defined hypoechoic mass in the accessories, acoustic attenuation and minimal Doppler flow signal, as well as pleural fluids or ascites. However, it can be easily misdiagnosed as a malignant ovarian tumor due to its lack of understanding. However, it should also be noted that fibroma can spread out of the ovary and adhere to the surrounding tissues in sporadic cases. In this case, the patients have a poor prognosis and infection complication with increased recurrence risk. The recurrence time can be more than 10 years after the operation. An improved understanding of the disease is helpful to avoid misdiagnosis and missed diagnosis. Abbreviations WT-1: Wilms tumor- 1 protein; Ki-67: marker of proliferation Ki-67; HPFs: high power fields; Declarations Acknowledgments None. Authors’ contributions Na T supervised the literature search and wrote the paper. Kai J provided the interesting case that we reported. Xiao Y and Jin Y performed immunohistochemical tests. Cheng L evaluated the histopathological images. Hong Z revised manuscript as well as guidance and editing throughout the writing process. All authors have read and approved the final manuscript. Funding Zunyi Science and Technology Bureau Science and technology cooperation （2018）85. Availability of data and materials As a case report, all data generated or analyzed are included in this article. Ethics approval and consent to participate Not applicable. Consent for publication Written informed consent was obtained from the patient for the publication of this case report. Competing interests The authors declare that they have no competing interests. References Chung BM, Park SB, Lee JB, Park HJ, Kim YS, Oh YJ. Magnetic resonance imaging features of ovarian fibroma, fibrothecoma, and thecoma. Abdom Imaging. 2015;40(5):1263-1272. Chen YJ, Hsieh CS, Eng HL, Huang CC. Ovarian fibroma in a 7-month-old infant: a case report and review of the literature. Pediatr Surg Int . 2004;20(11-12):894-897. Zhang Z, Wu Y, Gao J. CT diagnosis in the thecoma-fibroma group of the ovarian stromal tumors. Cell Biochem Biophys. 2015;71(2):937-943. Hogan ME, Andreotti R. Ovarian Fibroma Presenting With Meigs Syndrome. Ultrasound Q. 2015;31(3):215-218. Numanoglu C, Kuru O, Sakinci M, Akbayır O, Ulker V. Ovarian fibroma/fibrothecoma: retrospective cohort study shows limited value of risk of malignancy index score. Aust N Z J Obstet Gynaecol. 2013;53(3):287-292. Shen Y, Liang Y, Cheng X, Lu W, Xie X, Wan X. Ovarian fibroma/fibrothecoma with elevated serum CA125 level: A cohort of 66 cases. Medicine (Baltimore). 2018;97(34):e11926. Nemeth AJ, Patel SK. Meigs syndrome revisited. J Thorac Imaging. 2003;18(2):100-103. Cheng MH, Yen MS, Chao KC, Sheu BC, Wang PH. Differential diagnosis of gynecologic organ-related diseases in women presenting with ascites. Taiwan J Obstet Gynecol. 2008;47(4):384-390. Roth LM. Recent advances in the pathology and classification of ovarian sex cord-stromal tumors. Int J Gynecol Pathol. 2006;25(3):199-215. Mohammed SA, Kumar A. Meigs Syndrome. In: StatPearls. Treasure Island (FL): StatPearls Publishing; February 9, 2021. Torres Jiménez AR, Solís-Vallejo E, Céspedes-Cruz AI, Zeferino Cruz M, Rojas-Curiel EZ, Sánchez-Jara B. Tjalma syndrome (pseudo-pseudo Meigs') as initial manifestation of juvenile-onset systemic lupus erythematosus. Síndrome de Tjalma (pseudo-pseudo Meigs) como manifestación inicial de lupus eritematoso sistémico de inicio juvenil. Reumatol Clin (Engl Ed). 2019;15(5):e41-e43. Additional Declarations No competing interests reported. Cite Share Download PDF Status: Under Review Version 1 posted Editorial decision: Major revision 19 Feb, 2022 Reviews received at journal 17 Feb, 2022 Reviewers agreed at journal 15 Feb, 2022 Reviewers invited by journal 11 Feb, 2022 Editor assigned by journal 11 Feb, 2022 Submission checks completed at journal 23 Dec, 2021 First submitted to journal 22 Dec, 2021 You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-1374873","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":84996996,"identity":"47b22efc-d053-4de9-8111-1af23ee3a1aa","order_by":0,"name":"Na Tan","email":"","orcid":"","institution":"Affiliated Hospital of Zunyi Medical University","correspondingAuthor":false,"submittingAuthor":false,"prefix":"","firstName":"Na","middleName":"","lastName":"Tan","suffix":""},{"id":84996997,"identity":"9de5cb97-9532-4f75-8962-344c743c175b","order_by":1,"name":"Kai-yuan Jin","email":"","orcid":"","institution":"Affiliated Hospital of Zunyi Medical University","correspondingAuthor":false,"submittingAuthor":false,"prefix":"","firstName":"Kai-yuan","middleName":"","lastName":"Jin","suffix":""},{"id":84996998,"identity":"38d09b2f-4f5e-4edc-acdd-16efb29cb663","order_by":2,"name":"Xiao-rong Yang","email":"","orcid":"","institution":"Affiliated Hospital of Zunyi Medical University","correspondingAuthor":false,"submittingAuthor":false,"prefix":"","firstName":"Xiao-rong","middleName":"","lastName":"Yang","suffix":""},{"id":84996999,"identity":"e70d2863-6b28-4ab2-923e-ee6c1a6897f0","order_by":3,"name":"Cheng-fang Li","email":"","orcid":"","institution":"Affiliated Hospital of Zunyi Medical University","correspondingAuthor":false,"submittingAuthor":false,"prefix":"","firstName":"Cheng-fang","middleName":"","lastName":"Li","suffix":""},{"id":84997000,"identity":"ad025a94-f196-4f45-9077-b163f49221ad","order_by":4,"name":"Jin Yao","email":"","orcid":"","institution":"Affiliated Hospital of Zunyi Medical University","correspondingAuthor":false,"submittingAuthor":false,"prefix":"","firstName":"Jin","middleName":"","lastName":"Yao","suffix":""},{"id":84997001,"identity":"a0626b01-36d3-499f-ac81-b96c5986382e","order_by":5,"name":"Hong Zheng","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA+klEQVRIiWNgGAWjYDACCSBmbAAzGB+DRZiZG4jWwmzMwGAA1MJIvBY2abAWBgJa5Gc3H3v4dYdNnnx087bqgoo/0fztQC0/Krbh1MI451i6seyZtGLDO8fKbs84Y5A74zBjA2PPmds4tTBL5JhJS7YdTtw4I8fsNm+bQW4DUAszYxtuLWwS+d/gWopBWuYT0sIjkcMm+RGoZT7QOmaQlg2EtEhIpJlJM7alJW6QSCuW5jljnLsRqOUgPr/Iz0h+JvmzzSZx/ozkjZ95KuRy550/fPDBjwrcWsBBwAMkDA6A4wQCDuBVDwSMP0DWNSBpGQWjYBSMglGADACzX1ja+fQ6twAAAABJRU5ErkJggg==","orcid":"","institution":"Affiliated Hospital of Zunyi Medical University","correspondingAuthor":true,"submittingAuthor":false,"prefix":"","firstName":"Hong","middleName":"","lastName":"Zheng","suffix":""}],"badges":[],"createdAt":"2022-02-19 03:27:52","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-1374873/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-1374873/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":18527689,"identity":"5916292e-896e-492d-84cb-5789ca693a4a","added_by":"auto","created_at":"2022-02-23 15:46:07","extension":"jpg","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":200188,"visible":true,"origin":"","legend":"\u003cp\u003eA and B, CT coronal images through the 10th thoracic vertebrae and the pelvis demonstrate there were multiple irregular cystic and solid masses in bilateral adnexa (White small arrow). A large amount of effusion (white big arrowhead) is present within the bilateral thoracic cavity and pelvic and abdominal cavity. the uterus (circle 1), the bladder (circle 2).\u003c/p\u003e","description":"","filename":"Fig1.jpg","url":"https://assets-eu.researchsquare.com/files/rs-1374873/v1/25c5f00eb0f5a869b4d50c50.jpg"},{"id":18528463,"identity":"6436fba8-18f7-4d3a-bc95-9ffc54543f2a","added_by":"auto","created_at":"2022-02-23 15:49:07","extension":"jpg","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":71543,"visible":true,"origin":"","legend":"\u003cp\u003eLaparoscopic Intraoperative photograph, can be found bilateral ovarian fibroma (black cross) and ascites (black arrow).\u003c/p\u003e","description":"","filename":"Fig2.jpg","url":"https://assets-eu.researchsquare.com/files/rs-1374873/v1/42863f0c590759f8771953e8.jpg"},{"id":18527693,"identity":"4bbe03a5-55ea-47f2-acea-6252485c62c9","added_by":"auto","created_at":"2022-02-23 15:46:07","extension":"jpg","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":1023749,"visible":true,"origin":"","legend":"\u003cp\u003eA Histology: showed a large number of spindle cells or ovoid cells，Tumor cell nucleus is long fusiform. There is an obvious edema between cells (400×). B Immunohistochemistry: α-inhibin weakly positive (100×). C Immunohistochemistry: Vimentin positive (100×). D Immunohistochemistry: Reticular fiber positive (400×).\u003c/p\u003e","description":"","filename":"Fig3.jpg","url":"https://assets-eu.researchsquare.com/files/rs-1374873/v1/17a601884f360d6b06b65637.jpg"},{"id":18528465,"identity":"0c059ed1-9389-4429-ab68-c5a0bfa459af","added_by":"auto","created_at":"2022-02-23 15:49:10","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":379589,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-1374873/v1/16fc8389-6efe-430c-8712-5d3b94233219.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"\u003cp\u003eA Case of Death of Patient With Ovarian Fibroma Combined With Meigs Syndrome and Literature Review\u003c/p\u003e","fulltext":[{"header":"Introduction","content":"\u003cp\u003eA 46-year-old female was presented with recurrent abdominal distension and pain for 4 years. The symptoms worsened with vomiting for 1 month, and the weight loss was 5 kg recently. The diagnosis before admission: duodenal stenosis, pyloric obstruction? ovarian cancer? Observations by ultrasound: bilateral adnexal mass, pelvic and peritoneal effusion. Plain and enhanced CT scan of the abdomen (see Fig. 1): there were multiple irregular cystic and solid masses in bilateral adnexa, considered neoplastic lesions. It should think about the possibility of ovarian cancer. Moreover, a large amount of effusion was found in the bilateral thoracic cavity and pelvic and abdominal cavity. There was effusion in abdominal soft tissue. Female tumor-associated antigen = CA19-9 41.10 U/mL and CA125 56.5 U/mL (normally \u0026lt; 35 U/mL). Biochemical tests of ascites: total proteins 42.3 g/L, rivalta test (+). The clinical diagnosis: causes of abdominal distension: metastasis and invasion of ovarian cancer? pyloric obstruction? multiple abdominal metastases? Intraoperative observation: there exited adhesion with a hard texture between the middle segment of the greater omentum and lower margin of the right hepatic lobe and between the duodenal bulb and pyloric part. A large amount of pale blood fluid could be found under the left subphrenic space, right liver lobe and pelvic cavity. The bilateral ovaries showed nodular and beaded changes, with multiple nodules. The nodules had different sizes, with slightly hard quality and acceptable mobility. Intraoperative frozen sections: spindle cell tumor. It was considered the diagnosis of sex cord-stromal tumor. Subtotal gastrectomy and gastrojejunostomy + distal duodenal exclusion + complex enterolysis + bilateral accessory and hysterectomy were performed. The patient frequently vomited after the operation, and 1500 mL liquid was drained from the gastric tube. The patient had a fever and a CT scan of the chest showed increased pleural effusion. There were secondary coagulation dysfunction, sepsis and multiple organ failure.\u003c/p\u003e"},{"header":"Pathological Examination","content":"\u003cp\u003eObservation by the naked eye: the sizes of bilateral ovaries were 8\u0026times;6\u0026times;4 cm\u003csup\u003e3\u003c/sup\u003e and 7\u0026times;5\u0026times;2 cm\u003csup\u003e3\u003c/sup\u003e, respectively. The surface of the ovaries was smooth with multinodular and beaded changes. The size of the nodules varied, with the largest diameter of 3 cm (see Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003e). The surface section was off-white, with moderate hardness and solid structure. The ovaries were cystic locally and the thickness of the cyst wall was 0.1 cm with colloidal substances inside. There was no bleeding or necrosis.\u003c/p\u003e \u003cp\u003eMicroscopic examination (see Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003e): the ovarian structure disappeared. A large number of spindle cells or oval cells are arranged in feathery, braided, or whirl-like, occasionally storiform. The nuclei of tumor cells were long fusiform without mitotic figures. There was no lipid in the cytoplasm. The intercellular edema was prominent without mucoid degeneration, hyaline degeneration, calcification and ossification. The results of immunohistochemistry showed that tumor cells: Vimentin, WT-1, Calretinin (partial expression), Ki-67 (3% positive rate), α-inhibin (+), reticular fiber dyeing (+).\u003c/p\u003e \u003cp\u003ePathological examination: ovarian fibroma.\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eAccording to the definition of female reproductive system diseases proposed by WHO in 2020, ovarian fibroma is a benign pure stromal tumor composed of the spindle to oval fibroblasts like cells that produce collagen. Ovarian fibroma is the most common sex cord-stromal tumor, accounting for 4% of all ovarian tumors.\u003csup\u003e[\u003cspan class=\"CitationRef\"\u003e1\u003c/span\u003e]\u003c/sup\u003e It can occur at any age, but it is most common in middle age (average 48 years old). It seldom occurs before 30 years old and has also been reported in infants of 7 months old. \u003csup\u003e[\u003cspan class=\"CitationRef\"\u003e2\u003c/span\u003e]\u003c/sup\u003e Most of the tumors are unilateral and rarely bilateral. The tumor\u0026rsquo;s average size is 8 cm, about 1/3 of the tumors can be less than 3 cm. There was no specific clinical manifestation and most of them show pelvic or adnexal masses.\u003csup\u003e[\u003cspan class=\"CitationRef\"\u003e3\u003c/span\u003e]\u003c/sup\u003e\u003c/p\u003e\n\u003cp\u003eAbout 10%-15% of ovarian fibroma can be combined with pleural fluids or ascites, described as Meigs syndrome. It was reported by Meigs and Cassa in 1937. In 1954, Meigs Syndrome was defined as a benign ovarian fibroma or fibroid tumor with pleural fluids and ascites. The pleural fluids and ascites disappeared after tumor resection, and ascites recurred in tumors with a diameter \u0026gt; 10 cm. The possible cause of pleural fluids and ascites is that the fluid within tumors infiltrates the abdominal cavity through the serosal surface. It then enters into the unilateral or bilateral pleural cavity through the lymphatic vessels or the communication between the pleural cavity and the abdominal cavity.\u003csup\u003e[\u003cspan class=\"CitationRef\"\u003e4\u003c/span\u003e]\u003c/sup\u003e\u003c/p\u003e\n\u003cp\u003eThere are no specific markers for accurate diagnosis of ovarian fibroids/fibroids before surgery.\u003csup\u003e[\u003cspan class=\"CitationRef\"\u003e5\u003c/span\u003e]\u003c/sup\u003e CA125 level in serum increased in a few patients. Some studies suggested that increased CA125 level in serum might be related to some biochemical factors, such as elevated intraperitoneal pressure caused by mechanical stimulation of ascites to the peritoneum, or peritoneal mesothelial cells, which might be misdiagnosed as endometriosis or malignant ovarian tumor. \u003csup\u003e[\u003cspan class=\"CitationRef\"\u003e6\u003c/span\u003e]\u003c/sup\u003e In this case, the patient had an elevated CA125 and ascites before the operation, which was easily misdiagnosed as a malignant ovarian tumor.\u003c/p\u003e\n\u003cp\u003eAccording to the proportion of fibroblasts and collagen fibers, ovarian fibroma can be divided into cellular and fibrous types. Cellular fibroma is associated with ovarian rupture and extraovarian adhesions and has a risk of local recurrence. Besides, fibroma has some particular subtypes. Fibroma with a small amount of sex cord components refers to less than 10% tubules with different differentiation degrees or incomplete tubular structure of immature supporting cells in fibroma. Moreover, there are abundant tumor cells arranged closely with unclear cytoplasmic boundaries and fewer collagen components. The nuclear chromatin increases and is in round or oval with or without mild atypia, and mitotic rates are \u0026ge; 1-3/10 HPFs. In this case, it can be called cell-rich fibroma. Cell-rich fibroma commonly presents a benign clinical course but has low malignant potential and occasional local recurrence.\u003csup\u003e[\u003cspan class=\"CitationRef\"\u003e7\u003c/span\u003e, \u003cspan class=\"CitationRef\"\u003e8\u003c/span\u003e, \u003cspan class=\"CitationRef\"\u003e9\u003c/span\u003e]\u003c/sup\u003e\u003c/p\u003e\n\u003cp\u003eIt should be differentiated from the following diseases:\u003c/p\u003e\n\u003col\u003e\n\u003cli\u003e\n\u003cp\u003eThecoma: thecoma is another kind of pure stromal tumor. However, fibroma and thecaoma have overlapping morphological features. Some cases that are difficult to distinguish can be called fibrothecoma or follicular fibroma. Microscopically, several relatively isolated theca cells rich in lipid between fat spindle-shaped fibroblasts or fibroblasts, showing clump distribution. However, if the patient has no steroid hormone changes and the composition of theca cells in tumors is less than 10% with negative immunohistochemical inhibin, it is not recommended to be diagnosed as fibrothecoma but directly diagnosed as a fibroma. Theca cells in this patient are not rich in lipid and have loose fibrous stroma with prominent edema.\u003c/p\u003e\n\u003c/li\u003e\n\u003cli\u003e\n\u003cp\u003eMassive edema of the ovary: when fibroma shows obvious edema, it should be differentiated from this disease. Most of the patients with massive edema of the ovary are younger than 30 years old, and abdominal pain is the main clinical symptom. Ovaries have the normal structure, and the whole ovary is diffuse with edema, and sometimes luteinized cells can be found in the stroma. The patient, in this case, does not have ovaries with the normal structure.\u003c/p\u003e\n\u003c/li\u003e\n\u003cli\u003e\n\u003cp\u003eFibrosarcoma: the tumor cells with moderate to severe atypia are very abundant, arranged in braided shape, with eosinophilic cytoplasm and unclear boundaries. The nuclei are in round or fusiform shape, with abundant chromatin, prominent nucleolus. Mitotic images are common, with an average of \u0026gt; 4/10 HPFs. There usually are bleeding and necrosis complications. The transitional zone between fibroma and fibrosarcoma can be found in a few tumors. Fibrosarcoma has a poor prognosis and a few patients had been reported to survive more than 5 years.\u003c/p\u003e\n\u003c/li\u003e\n\u003cli\u003e\n\u003cp\u003eThe identification of Meigs Syndrome:\u003c/p\u003e\n\u003c/li\u003e\n\u003c/ol\u003e\n\u003cp\u003ePseudo Meigs Syndrome: in addition to ovarian fibroma, ascites and pleural fluids occur in patients with other benign or malignant tumors in the pelvis or abdomen.\u003csup\u003e[\u003cspan class=\"CitationRef\"\u003e10\u003c/span\u003e]\u003c/sup\u003e\u003c/p\u003e\n\u003cp\u003ePseudo-Pseudo Meigs Syndrome: it is also known as Tajalma Syndrome. It can be found in patients with systemic lupus erythematosus accompanied by ascites, pleural fluids, and elevated CA125 level in serum.\u003csup\u003e[\u003cspan class=\"CitationRef\"\u003e11\u003c/span\u003e]\u003c/sup\u003e\u003c/p\u003e\n\u003cp\u003eThe patient had ovarian fibroma with pleural fluids and ascites, accompanied by slightly elevated CA125. There were no other tumors or systemic lupus erythematosus. After bilateral accessory and hysterectomy, the pleural fluids and ascites disappeared temporarily, consistent with Meigs Syndrome. The subsequent recurrence of pleural fluids and ascites was due to severe infection rather than the disease itself.\u003c/p\u003e\n\u003cp\u003eBased on the ultrasound imaging, the typical ovarian fibroma can be manifested as a well-defined hypoechoic mass in the accessories, acoustic attenuation and minimal Doppler flow signal, as well as pleural fluids or ascites. However, it can be easily misdiagnosed as a malignant ovarian tumor due to its lack of understanding. However, it should also be noted that fibroma can spread out of the ovary and adhere to the surrounding tissues in sporadic cases. In this case, the patients have a poor prognosis and infection complication with increased recurrence risk. The recurrence time can be more than 10 years after the operation. An improved understanding of the disease is helpful to avoid misdiagnosis and missed diagnosis.\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cp\u003eWT-1: Wilms tumor- 1 protein; Ki-67: marker of proliferation Ki-67; HPFs: high power fields;\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eAcknowledgments\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNone.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAuthors\u0026rsquo; contributions\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNa T supervised the literature search and wrote the paper. Kai J provided the interesting case that we reported. Xiao Y and Jin Y performed immunohistochemical tests. Cheng L evaluated the histopathological images. Hong Z revised manuscript as well as guidance and editing throughout the writing process. All authors have read and approved the final manuscript.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFunding\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eZunyi Science and Technology Bureau\u003c/p\u003e\n\u003cp\u003eScience and technology cooperation （2018）85.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAvailability of data and materials\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eAs a case report, all data generated or analyzed are included in this article.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eEthics approval and consent to participate\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNot applicable.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent for publication\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eWritten informed consent was obtained from the patient for the publication of this case report.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCompeting interests\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe authors declare that they have no competing interests.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n \u003cli\u003eChung BM, Park SB, Lee JB, Park HJ, Kim YS, Oh YJ. Magnetic resonance imaging features of ovarian fibroma, fibrothecoma, and thecoma. Abdom Imaging. 2015;40(5):1263-1272.\u003c/li\u003e\n \u003cli\u003eChen YJ, Hsieh CS, Eng HL, Huang CC. Ovarian fibroma in a 7-month-old infant: a case report and review of the literature. \u003cem\u003ePediatr Surg Int\u003c/em\u003e. 2004;20(11-12):894-897.\u003c/li\u003e\n \u003cli\u003eZhang Z, Wu Y, Gao J. CT diagnosis in the thecoma-fibroma group of the ovarian stromal tumors. Cell Biochem Biophys. 2015;71(2):937-943.\u003c/li\u003e\n \u003cli\u003eHogan ME, Andreotti R. Ovarian Fibroma Presenting With Meigs Syndrome. Ultrasound Q. 2015;31(3):215-218.\u003c/li\u003e\n \u003cli\u003eNumanoglu C, Kuru O, Sakinci M, Akbayır O, Ulker V. Ovarian fibroma/fibrothecoma: retrospective cohort study shows limited value of risk of malignancy index score. Aust N Z J Obstet Gynaecol. 2013;53(3):287-292.\u003c/li\u003e\n \u003cli\u003eShen Y, Liang Y, Cheng X, Lu W, Xie X, Wan X. Ovarian fibroma/fibrothecoma with elevated serum CA125 level: A cohort of 66 cases. Medicine (Baltimore). 2018;97(34):e11926.\u003c/li\u003e\n \u003cli\u003eNemeth AJ, Patel SK. Meigs syndrome revisited. J Thorac Imaging. 2003;18(2):100-103.\u003c/li\u003e\n \u003cli\u003eCheng MH, Yen MS, Chao KC, Sheu BC, Wang PH. Differential diagnosis of gynecologic organ-related diseases in women presenting with ascites. Taiwan J Obstet Gynecol. 2008;47(4):384-390.\u003c/li\u003e\n \u003cli\u003eRoth LM. Recent advances in the pathology and classification of ovarian sex cord-stromal tumors. Int J Gynecol Pathol. 2006;25(3):199-215.\u003c/li\u003e\n \u003cli\u003eMohammed SA, Kumar A. Meigs Syndrome. In: StatPearls. Treasure Island (FL): StatPearls Publishing; February 9, 2021.\u003c/li\u003e\n \u003cli\u003eTorres Jim\u0026eacute;nez AR, Sol\u0026iacute;s-Vallejo E, C\u0026eacute;spedes-Cruz AI, Zeferino Cruz M, Rojas-Curiel EZ, S\u0026aacute;nchez-Jara B. Tjalma syndrome (pseudo-pseudo Meigs\u0026apos;) as initial manifestation of juvenile-onset systemic lupus erythematosus. S\u0026iacute;ndrome de Tjalma (pseudo-pseudo Meigs) como manifestaci\u0026oacute;n inicial de lupus eritematoso sist\u0026eacute;mico de inicio juvenil. Reumatol Clin (Engl Ed). 2019;15(5):e41-e43.\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":true,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"diagnostic-pathology","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"dpat","sideBox":"Learn more about [Diagnostic Pathology](http://diagnosticpathology.biomedcentral.com)","snPcode":"13000","submissionUrl":"https://submission.nature.com/new-submission/13000/3","title":"Diagnostic Pathology","twitterHandle":"@OncoBioMed","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"BMC/SO AJ","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"ovarian fibroma, Meigs Syndrome","lastPublishedDoi":"10.21203/rs.3.rs-1374873/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-1374873/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003eOvarian fibroma is the most common benign pure stromal tumor. It has no specific clinical manifestation, most of which are pelvic or adnexal masses. 10%-15% of cases with hydrothorax or ascites, after tumor resection, hydrothorax and ascites disappear, known as Meigs Syndrome. The elevated level of CA125 in a few patients was easily misdiagnosed as ovarian malignant tumor. A case of bilateral Ovarian fibroma associated with Meigs Syndrome is reported and the literature is reviewed in order to improve the understanding of the changes and avoid misdiagnosis.\u003c/p\u003e","manuscriptTitle":"A Case of Death of Patient With Ovarian Fibroma Combined With Meigs Syndrome and Literature Review","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2022-02-23 15:46:05","doi":"10.21203/rs.3.rs-1374873/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Major revision","date":"2022-02-19T16:13:44+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2022-02-17T19:00:14+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"023fb6d0-26e0-4f2a-8f31-609afe9c3eaa","date":"2022-02-15T17:56:14+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2022-02-12T02:33:27+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2022-02-11T11:29:57+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2021-12-23T16:47:19+00:00","index":"","fulltext":""},{"type":"submitted","content":"Diagnostic Pathology","date":"2021-12-23T00:29:21+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"diagnostic-pathology","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"dpat","sideBox":"Learn more about [Diagnostic Pathology](http://diagnosticpathology.biomedcentral.com)","snPcode":"13000","submissionUrl":"https://submission.nature.com/new-submission/13000/3","title":"Diagnostic Pathology","twitterHandle":"@OncoBioMed","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"BMC/SO AJ","inReviewEnabled":true,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"d5e5fa7a-fd65-4935-88e0-4289ceb5b8ea","owner":[],"postedDate":"February 23rd, 2022","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"under-review","subjectAreas":[],"tags":[],"updatedAt":"2022-09-15T22:29:09+00:00","versionOfRecord":[],"versionCreatedAt":"2022-02-23 15:46:05","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-1374873","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-1374873","identity":"rs-1374873","version":["v1"]},"buildId":"ehx78VzkSd0WSzXnipQa-","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}