Teratoma combined with struma ovarii and sarcomatoid carcinoma：A case report and review of the literature

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Whether it's struma ovarii or ovarian sarcomatoid carcinoma, the incidence of both is extremely low and rarely reported, so there are no clear guidelines for either disease and to our knowledge this may be the first case of combined occurrence of both. Therefore, this report describes its clinical manifestations, diagnosis and treatment, analyzes the pathogenesis, and summarizes the previous literature in the hope that it can be helpful to other tumor-related medical personnel and provide material support for the formation of guidelines in this disease. teratoma struma ovarii ovarian sarcomatoid carcinoma transformation theory Figures Figure 1 Figure 2 Figure 3 Figure 4 Figure 5 Figure 6 Figure 7 Figure 8 Introduction Ovarian teratoma is a kind of ovarian germ cell tumor with mainly benign lesions, accounting for about 15% of the total number of primary ovarian tumors, and its malignant change rate is only 0.2% ~ 2%, and mainly squamous cell carcinoma [ ] . Struma Ovarii (SO) is a special pathological type of teratoma differentiated from a single germ layer, accounting for about 2% ~ 3% of ovarian teratoma and 0.1% ~ 0.5% of all ovarian tumors [ ] , incorporation with sarcomatoid carcinoma is even rarer. Therefore, the clinical diagnosis, treatment plan, and prognosis evaluation of Struma Ovarii with sarcomatoid carcinoma are still unclear, and it is difficult to form a convincing expert consensus or guidelines for gynecologic oncologists. This paper reported a case of ovarian teratoma combined with struma ovarii and sarcomatoid carcinoma, summarized its clinical characteristics and treatment methods, and combined with the literature to improve the relevant knowledge of the disease, and provide reference for the diagnosis, treatment and prognosis. Case presentation A 67-year-old woman who was admitted to the hospital with "the lower abdomen was unwell for more than half a year, and the pelvic mass was found for 5 days". In May 2023, she felt abdominal discomfort and occasionally had abdominal pain. She took oral antibiotics for more than half a month, and abdominal pain was slightly relieved. In June 2023, there was more pain in the lower abdomen than before. On December 18, 2023, ultrasound at local hospital suggested multiple pelvic masses; pelvic MRI showed the right accessory area mass (9.8 × 6.3 ㎝ confounding signal), indicating the possibility of teratoma. On December 25, 2023, she came to our hospital for further treatment. The patient has no fever recently, and her diet, sleep, urinary and bowel function are normal. She had regular menstruation previously, with natural menopause in 51 years old, G2A0P2, VD. Past medical history and family history of tumor were denied. Physical examination after admission: vital signs were stable, generally in good condition, and no abnormal physical development. There was no palpable enlargement of superficial lymph nodes throughout the body and no abnormalities on cardiopulmonary auscultation. The abdomen was flat without tenderness and rebound pain, the liver and spleen were not palpable under the ribs, negative shifting dullness. Physical examination of gynecology: vulva development is normal, left labia and perineal combined skin thickening, hypopigmentation. Vaginal patency, postpartum cervix, smooth posterior lip, and erosion changes in the anterior lip. The uterus was unsatisfactory on palpation, no mass was touched in the left appendage area, and the mass in the right appendage area was about 9 × 7× 7cm in size. The rectal mucosa was smooth and nodules were not touched in the uterine rectal fossa. Auxiliary examination: the routine blood test and liver and kidney function results were normal. Tumor markers were of normal range: CA199 3.51 U / mL; AFP 4.33 ng/mL; CEA 2.08 ng/mL; CA125 26.40 U / mL; HE422.70 pmol / L. HPV: (-);TCT༚NILM. ECG: Normal. Cardiac ultrasound: in the resting state, the left ventricular overall systolic function is normal. Pulmonary function test: moderate restricted ventilation dysfunction, mild ventilation dysfunction, and reduced small airway function. CT of the lung and abdomen: irregular mass on the right side of the pelvic cavity, ranging about 9.1cm×6.7cm, mixed density, fat density and calcification, and enhancement of soft tissue composition; no enlarged lymph nodes in the retroperitoneal area (Fig. 1). Diagnosis and treatment: after a comprehensive examination, the preliminary diagnosis of pelvic mass (malignancy cannot be excluded). After evaluation of anesthesiologists of our hospital, there were no absolute surgical contraindications, and the exploratory laparotomy was performed under general anesthesia on December 27, 2023. Intraoperative observation: no ascites, no abnormality in the upper abdomen. The right ovary was replaced by a mass, about 10 × 8 × 7 ㎝ in size, and the right fallopian tube was attached to the mass; the appearance of the left ovary and fallopian tube was normal. No significant abnormalities were observed in the uterus and greater omentum appearance. The peritoneum of the bladder wall layer was adhered to the right ovarian mass with an area of about 4 × 3 ㎝. After separation, the abnormal peritoneal membrane was completely removed and sent to intraoperative cryopathology. The result of frozen pathology showed that the heterogeneous cells were seen in the fiber stroma, which was considered carcinoma. The right ovarian vessels were isolated by high ligation, the right broad ligament was freed, the right utero-ovarian ligament was isolated, and the right ovary and fallopian tube were completely removed and sent for intraoperative cryopathology. The result was (right attachment) struma ovarii, and the diffuse distribution of heterogeneous cells in some areas was malignant. Based on the cryopathology results, the intraoperative diagnosis of ovarian cancer was stage IIB, according to the 2023 NCCN guidelines of ovarian cancer [ ] . Then, the uterus and left ovary fallopian tube, greater omentum were complete resected, with peritoneal multipoint biopsy. The lymph nodes in the region of the right and left obturator foramen, internal and external iliac, common iliac, and para-abdominal aorta were not found to be enlarged on exploration and were left untreated. After the operation, the patient safely returned to the ward. After surgery, the gross specimen was examined as follows (Fig. 2A); upon dissection of the mass, a cystic-solid component was visible within the right ovary, with contents including oil, hair, bone structure, and thyroid components (Fig. 2B). Microscopic pathological morphological features and immunohistochemical results: The morphology is dominated by a teratoma component with polygonal differentiation and prominent zones of heterogeneous spindle cells, which has a clear line between the two (Fig. 3), and tumor cell infiltration could be seen under the peritoneum of the bladder wall (Fig. 4). The teratoma component contains mature thyroid follicular structures (a variable amount of eosinophilic colloids can be seen in the cavity, the follicular epithelium is a single layer of low column or cubic form), and it accounts for > 50% of tumors, and differentiated and mature squamous epithelium and skin appendages (sebaceous gland and hair follicle structures) can also be found;the spindle cell region is characterized by significant cellular heterogeneity, deeply stained nuclei, and eosinophilic cytoplasm. Immunohistochemical results were CK (+), Vimentin (+), Ki67 (80% +), S-100 (-), Desmin (-), MyoD 1 (-), P53 (+) / mutant, P40 (-), Sall-4 (-), Pax-8 (-), SMA minority cells (+), it suggests that this region has epithelial and mesenchymal origin of biphasic differentiation and does not support differentiation into nerve, striated muscle, female germ cell tumors, and squamous cell carcinoma (Fig. 5–8). Postoperative treatment: TC chemotherapy is recommended. The patient is currently on chemotherapy with no adverse reactions above II°. Discussion Ovarian teratoma was classified into mature teratoma (benign) and immature teratoma (malignant) according to their pathological nature, 95% of which were mature teratoma [ ] . Early symptoms are not obvious, mainly to the physical examination found, surgery is the main treatment means currently [ ] . SO is a highly specific monoembryonic teratoma, which diagnostic criteria are microscopic thyroid tissue composition > 50%, or thyroid tissue < 50% but with significant hyperthyroidism, or visually recognizable thyroid tissue in a mature teratoma [ ] . SO has predominantly unilateral, left-sided onset, with right-sided onset in slightly older patients and bilateral onset in 8% of patients [ ] . The right-sided onset of the disease in this elderly patient is consistent with the literature. The clinical incidence of SO is very low, lacking typical clinical symptoms. Most of them are found by physical examination or with abdominal mass or abdominal pain. However, some studies have reported that about one third of clinical patients have "Meigs syndrome" [ ] . Based on the high incidence of pleural effusion and ascites and high level of CA125 expression in struma ovarii, some experts suggest that pleural effusion, ascites, and elevated CA125 should be included in the differential diagnosis of pelvic mass and struma ovarii [ ] . However, CA125 will increase in both benign and malignant cases, and will not continue to rise even in malignant cases. It is speculated that the increase in CA125 is not a direct result of the existence of the tumor itself, but rather a side effect of ascites [ ] . It is worth noting that although SO is a tumor composed of thyroid tissue, only 8% of the patients have hyperthyroidism [ ] . In this case, thyroid function was not tested because the SO was not diagnosed preoperatively, and the patient did not show symptoms of hyperthyroidism. This suggests that thyroid tissue of SO does not have the full function of synthetic thyroid hormone in most cases, but studies show that thyroglobulin can be used as a diagnostic reference for SO [ ] . Because of the clinical features of SO are also similar to those of ovarian malignancies, so preoperative imaging diagnosis becomes more important to distinguish ovarian cancer and avoid cancer-type surgery (eg, bilateral salpingectomy, hysterectomy). Ultrasound is the first choice for evaluation of ovarian masses during imaging studies [ ] . However, the ultrasound findings of struma ovarii are ambiguous, usually manifests as a multilocular cystic ovarian mass with solid components of various amounts, the ultrasound typically demonstrates these non-specific heterogeneous solid cystic features [ ] . The SO has features overlapping with those of malignant ovarian epithelial tumors, though they both present as a unilateral complex adnexal mass often associated with ascites, or as multi-cystic mass with solid components and multiple cystic locules, usually including teratoma components. Familiarity with the "doughnut sign" and the "fat layering sign" can help in the differential diagnosis, but it is possible to overestimate the malignancy of SO [ ] . As SO is an uncommon tumor, unlike the most common types of teratoma, does not demonstrate lipid material on either CT or MRI [ ] , but 123I or 131I scintigraphy is useful for diagnosing a hyper functional SO [ ] . Because SO is difficult to make a definitive preoperative diagnosis, postoperative paraffin pathology combined with immunohistochemistry is still the ultimate basis for diagnosis. In addition to thyroid tissue, there was obvious mature teratoma tissue [ ] , typical presentation: multilocular, uneven wall thickness, yellow and grayish-red jelly-like contents; immunohistochemical features: TTF 1 (+), TG (+), PAX-8 (+), CK7 (+), and Muc-1 (+). Due to the low incidence of SO and no clear diagnosis and treatment guidelines, surgery is still the main treatment modality. Due to its ultrasound morphology, which is quite similar to that of malignant ovarian carcinoma, most SO cases are often operated on with laparotomy, involving either ovariotomy or oophorectomy [ ] . For malignant struma ovarii, radioactive iodine supplementation after surgery is effective in preventing metastasis or recurrence [ ] . In addition to teratoma and SO of the pathological components, some malignant cells also be found which have significant spindle shape changes, including both epithelial markers and sarcoma markers. At first, we thought it was carcinosarcoma. However, carcinosarcoma needs to determine the components of carcinoma, such as adenocarcinoma, squamous carcinoma. Sarcoma also needs to identify the components, such as leiomyosarcoma, liposarcoma. Which has no clear carcinoma and sarcoma components named sarcomatoid carcinoma (SC) [ ] . The specific origin of the cancerous and sarcomatous components could not be determined in this case. The protein P40, which characterizes the most common squamous carcinoma of teratoma malignancy, is negative. But it expressed both epithelial (CK) and mesenchymal component (Vimentin) markers, so pathologists prefer to diagnose sarcomatoid carcinoma. SC is also known as spindle cell carcinoma, where the epithelial carcinoma component determines the biological behavior of metastasis, and the sarcoma component determines the prognosis. Usually, sarcomatoid tissue accounts for more than 50% of malignant tissues [ ] . In addition, it is necessary to perform immunohistochemistry to confirm the presence of epithelial components or sarcomatoid tissue surrounded by epithelial tissue that has heavy heterogeneous proliferation of carcinoma in situ. Otherwise, it should be differentiated from sarcoma, so as to avoid misdiagnosis when the proportion of sarcoma components in SC tissue is too large [ ] . Whether it is carcinosarcoma or sarcomatoid carcinoma, for germ cell tumors, which may have the characteristics of cell stemness, there are currently three theories of its formation mechanism: collision theory, combinatorial theory and transformation theory. "collision theory" believes that these two types of diseases have two components: epithelial cancer and sarcomatoid stroma, so it is proposed that tumor cells are biclonal, epithelial cancer components and sarcoma components are derived from two types of stem cells, evolve independently, and then collide; the "combination theory" suggests that a stem cell precursor in the early bidirectional differentiation into epithelial carcinoma components and sarcoma components; at present, the most recognized "transformation theory" proposes that the sarcoma component comes from carcinoma cells, or the original stem cells is differentiated into one kind of cell and re-differentiated to form a second cell. Its essence is a special type of cancer, and the sarcomatoid component is only the transformation of cancer component [ ] . A recent study tested RNA for cancer and sarcoma components in 18 patients with ovarian carcinosarcoma (OCS) found that the cancer component in OCS was more mesenchymal compared to epithelial ovarian cancer, supporting the conjecture that the sarcoma component is transformed through epithelial-mesenchymal cancer component. In this case, CK (+) and Vim (+) suggested that the spindle cell area was characterized by biphasic differentiation characteristics of epithelial and mesenchymal origin, which also tended to support the "transformation theory" [ ] . Although sarcomatoid carcinoma has been found in several organs, including the breast, bladder and kidney [ − ] , but there are few clinical reports and studies on ovarian sarcomatoid carcinoma (OSC), and only a few documents on PubMed, and most of them are case reports [ , ] .There are no standard guidelines for their diagnosis and treatment currently. Combined with the existing literature and our clinical experience, the early symptoms of OSC are not obvious, and they are usually found in physical examination or abdominal pain, abdominal distension and other manifestations. Tumor markers generally increase in CA125, and then the pelvic mass is found by imaging examination. Ultrasound cannot differentiate ovarian sarcomatoid carcinoma from other poorly differentiated ovarian cancers. CT can provide the condition around the lesion and the presence of peritoneal and lymph node metastasis, providing an important basis for clinical staging. MRI shows mixed image of hemorrhage, cyst or necrosis [ ] . Due to the difficulty of preoperative diagnosis of OSC, it is finally determined by postoperative pathological examination, as a result the condition has often progressed to advanced stages when be identified. There is no direct relevant literature for evaluation of prognosis, which can refer to the latest literature report of ovarian carcinosarcoma, which 5-year survival rate is 29.8%, and the median survival period is 16 to 24 months [ ] . Currently, the treatment of OSC is based on cytoreductive surgery combined with platinum-based chemotherapy, similar to ovarian carcinosarcoma or epithelial carcinoma [ ] . Among other treatments, radiotherapy is not recommended, and the efficacy of targeted therapy and immune checkpoint inhibitors for OSC is still controversial and needs more studies [ , ] . Aoki M et al found that the loss of expression of the ARID1A gene and the same PIK3CA mutation in different tissues, proving the monoclonality of sarcomatoid carcinoma and also providing evidence to support the transformation theory [ ] . According to the monoclonality of the disease, individual treatment options can be selected for the mutation gene to improve the survival rate and improve the quality of life for patients. Limitations (1) Although the patient was initially diagnosed as teratoma by color ultrasound and MRI, the patient had no history of thyroid disease or hyperthyroidism symptoms. Therefore, the relevant test was not carried out, and the struma ovarii was not diagnosed before surgery; (2) Up to now, the observation time is short, and the final survival and outcome still need to wait; (3) Genetic testing should be helpful to the diagnosis and treatment of this disease, but because its consumption is not covered by the Chinese medical insurance, so not be given to patients. In the future, we will apply for funding for genetic testing to find more clues about the pathogenesis from the genetic level. Abbreviations SO Struma Ovarii MRI Magnetic resonance imaging VD Vaginal delivery SC Sarcomatoid carcinoma OCS Ovarian carcinosarcoma OSC Ovarian sarcomatoid carcinoma Declarations Ethics approval and consent to participate Not applicable. Consent for publication This study obtained informed consent from the patient. Availability of data and materials Not applicable. Competing interests The authors declare no conflict of interest. Funding Not applicable. Authors' contributions H wrote the main manuscript text and was a surgical participant; D and S are surgical participants; J Managed the patient; B provide pathological diagnosis and specimen pictures; Y revised manuscript, the main participant in the surgery. Acknowledgments Not applicable. References Gadducci A, Guerrieri ME, Cosio S. Squamous cell carcinoma arising from mature cystic teratoma of the ovary: A challenging question for gynecologic oncologists. Crit Rev Oncol Hematol. 2019 Jan;133:92-98. Devi P, Aghighi M, Mikhail N. 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Ovarian teratoid carcinosarcoma with a PIK3CA mutation: a case report and review of the literature. Int Cancer Conf J. 2022 Aug 8;11(4):231-237. Additional Declarations No competing interests reported. Cite Share Download PDF Status: Published Journal Publication published 14 Sep, 2024 Read the published version in BMC Women's Health → Version 1 posted Editorial decision: Revision requested 12 Aug, 2024 Reviewers agreed at journal 11 Aug, 2024 Reviews received at journal 11 Aug, 2024 Reviewers agreed at journal 11 Aug, 2024 Reviews received at journal 10 Aug, 2024 Reviewers agreed at journal 10 Aug, 2024 Reviewers agreed at journal 10 Aug, 2024 Reviews received at journal 09 Aug, 2024 Reviews received at journal 09 Aug, 2024 Reviewers agreed at journal 09 Aug, 2024 Reviews received at journal 09 Aug, 2024 Reviews received at journal 09 Aug, 2024 Reviewers agreed at journal 09 Aug, 2024 Reviewers agreed at journal 09 Aug, 2024 Reviewers agreed at journal 09 Aug, 2024 Reviewers agreed at journal 09 Aug, 2024 Reviewers agreed at journal 09 Aug, 2024 Reviewers agreed at journal 09 Aug, 2024 Reviewers agreed at journal 09 Aug, 2024 Editor invited by journal 09 Aug, 2024 Reviewers invited by journal 07 Jun, 2024 Editor assigned by journal 22 May, 2024 Submission checks completed at journal 22 May, 2024 First submitted to journal 21 May, 2024 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. 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legend\u003c/p\u003e","description":"","filename":"7.png","url":"https://assets-eu.researchsquare.com/files/rs-4453757/v1/9c0de30770d7de7c73dedcbe.png"},{"id":57874094,"identity":"9d84c138-2f03-41a4-9de7-c757addeaedb","added_by":"auto","created_at":"2024-06-06 18:44:45","extension":"png","order_by":8,"title":"Figure 8","display":"","copyAsset":false,"role":"figure","size":3228184,"visible":true,"origin":"","legend":"\u003cp\u003eSee image above for figure legend\u003c/p\u003e","description":"","filename":"8.png","url":"https://assets-eu.researchsquare.com/files/rs-4453757/v1/f5150f247fe80a01956f8949.png"},{"id":64619707,"identity":"fcb79e36-b321-445c-a439-13c88db91758","added_by":"auto","created_at":"2024-09-16 16:16:56","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":30808651,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-4453757/v1/2cae99d6-d559-43c4-bda0-c9899fd8a21c.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Teratoma combined with struma ovarii and sarcomatoid carcinoma：A case report and review of the literature","fulltext":[{"header":"Introduction","content":"\u003cp\u003eOvarian teratoma is a kind of ovarian germ cell tumor with mainly benign lesions, accounting for about 15% of the total number of primary ovarian tumors, and its malignant change rate is only 0.2% ~ 2%, and mainly squamous cell carcinoma\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn1\" id=\"#FNLinkFn1\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. Struma Ovarii (SO) is a special pathological type of teratoma differentiated from a single germ layer, accounting for about 2% ~ 3% of ovarian teratoma and 0.1% ~ 0.5% of all ovarian tumors\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn2\" id=\"#FNLinkFn2\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e, incorporation with sarcomatoid carcinoma is even rarer. Therefore, the clinical diagnosis, treatment plan, and prognosis evaluation of Struma Ovarii with sarcomatoid carcinoma are still unclear, and it is difficult to form a convincing expert consensus or guidelines for gynecologic oncologists. This paper reported a case of ovarian teratoma combined with struma ovarii and sarcomatoid carcinoma, summarized its clinical characteristics and treatment methods, and combined with the literature to improve the relevant knowledge of the disease, and provide reference for the diagnosis, treatment and prognosis.\u003c/p\u003e"},{"header":"Case presentation","content":"\u003cp\u003eA 67-year-old woman who was admitted to the hospital with \"the lower abdomen was unwell for more than half a year, and the pelvic mass was found for 5 days\". In May 2023, she felt abdominal discomfort and occasionally had abdominal pain. She took oral antibiotics for more than half a month, and abdominal pain was slightly relieved. In June 2023, there was more pain in the lower abdomen than before. On December 18, 2023, ultrasound at local hospital suggested multiple pelvic masses; pelvic MRI showed the right accessory area mass (9.8 \u0026times; 6.3 ㎝ confounding signal), indicating the possibility of teratoma. On December 25, 2023, she came to our hospital for further treatment. The patient has no fever recently, and her diet, sleep, urinary and bowel function are normal. She had regular menstruation previously, with natural menopause in 51 years old, G2A0P2, VD. Past medical history and family history of tumor were denied.\u003c/p\u003e \u003cp\u003ePhysical examination after admission: vital signs were stable, generally in good condition, and no abnormal physical development. There was no palpable enlargement of superficial lymph nodes throughout the body and no abnormalities on cardiopulmonary auscultation. The abdomen was flat without tenderness and rebound pain, the liver and spleen were not palpable under the ribs, negative shifting dullness. Physical examination of gynecology: vulva development is normal, left labia and perineal combined skin thickening, hypopigmentation. Vaginal patency, postpartum cervix, smooth posterior lip, and erosion changes in the anterior lip. The uterus was unsatisfactory on palpation, no mass was touched in the left appendage area, and the mass in the right appendage area was about 9 \u0026times; 7\u0026times; 7cm in size. The rectal mucosa was smooth and nodules were not touched in the uterine rectal fossa.\u003c/p\u003e \u003cp\u003eAuxiliary examination: the routine blood test and liver and kidney function results were normal. Tumor markers were of normal range: CA199 3.51 U / mL; AFP 4.33 ng/mL; CEA 2.08 ng/mL; CA125 26.40 U / mL; HE422.70 pmol / L. HPV: (-);TCT༚NILM. ECG: Normal. Cardiac ultrasound: in the resting state, the left ventricular overall systolic function is normal. Pulmonary function test: moderate restricted ventilation dysfunction, mild ventilation dysfunction, and reduced small airway function. CT of the lung and abdomen: irregular mass on the right side of the pelvic cavity, ranging about 9.1cm\u0026times;6.7cm, mixed density, fat density and calcification, and enhancement of soft tissue composition; no enlarged lymph nodes in the retroperitoneal area (Fig.\u0026nbsp;1).\u003c/p\u003e \u003cp\u003eDiagnosis and treatment: after a comprehensive examination, the preliminary diagnosis of pelvic mass (malignancy cannot be excluded). After evaluation of anesthesiologists of our hospital, there were no absolute surgical contraindications, and the exploratory laparotomy was performed under general anesthesia on December 27, 2023. Intraoperative observation: no ascites, no abnormality in the upper abdomen. The right ovary was replaced by a mass, about 10 \u0026times; 8 \u0026times; 7 ㎝ in size, and the right fallopian tube was attached to the mass; the appearance of the left ovary and fallopian tube was normal. No significant abnormalities were observed in the uterus and greater omentum appearance.\u003c/p\u003e \u003cp\u003eThe peritoneum of the bladder wall layer was adhered to the right ovarian mass with an area of about 4 \u0026times; 3 ㎝. After separation, the abnormal peritoneal membrane was completely removed and sent to intraoperative cryopathology. The result of frozen pathology showed that the heterogeneous cells were seen in the fiber stroma, which was considered carcinoma. The right ovarian vessels were isolated by high ligation, the right broad ligament was freed, the right utero-ovarian ligament was isolated, and the right ovary and fallopian tube were completely removed and sent for intraoperative cryopathology. The result was (right attachment) struma ovarii, and the diffuse distribution of heterogeneous cells in some areas was malignant. Based on the cryopathology results, the intraoperative diagnosis of ovarian cancer was stage IIB, according to the 2023 NCCN guidelines of ovarian cancer \u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn3\" id=\"#FNLinkFn3\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. Then, the uterus and left ovary fallopian tube, greater omentum were complete resected, with peritoneal multipoint biopsy. The lymph nodes in the region of the right and left obturator foramen, internal and external iliac, common iliac, and para-abdominal aorta were not found to be enlarged on exploration and were left untreated. After the operation, the patient safely returned to the ward.\u003c/p\u003e \u003cp\u003eAfter surgery, the gross specimen was examined as follows (Fig.\u0026nbsp;2A); upon dissection of the mass, a cystic-solid component was visible within the right ovary, with contents including oil, hair, bone structure, and thyroid components (Fig.\u0026nbsp;2B).\u003c/p\u003e \u003cp\u003eMicroscopic pathological morphological features and immunohistochemical results: The morphology is dominated by a teratoma component with polygonal differentiation and prominent zones of heterogeneous spindle cells, which has a clear line between the two (Fig.\u0026nbsp;3), and tumor cell infiltration could be seen under the peritoneum of the bladder wall (Fig.\u0026nbsp;4). The teratoma component contains mature thyroid follicular structures (a variable amount of eosinophilic colloids can be seen in the cavity, the follicular epithelium is a single layer of low column or cubic form), and it accounts for \u0026gt;\u0026thinsp;50% of tumors, and differentiated and mature squamous epithelium and skin appendages (sebaceous gland and hair follicle structures) can also be found;the spindle cell region is characterized by significant cellular heterogeneity, deeply stained nuclei, and eosinophilic cytoplasm. Immunohistochemical results were CK (+), Vimentin (+), Ki67 (80% +), S-100 (-), Desmin (-), MyoD 1 (-), P53 (+) / mutant, P40 (-), Sall-4 (-), Pax-8 (-), SMA minority cells (+), it suggests that this region has epithelial and mesenchymal origin of biphasic differentiation and does not support differentiation into nerve, striated muscle, female germ cell tumors, and squamous cell carcinoma (Fig.\u0026nbsp;5\u0026ndash;8).\u003c/p\u003e \u003cp\u003ePostoperative treatment: TC chemotherapy is recommended. The patient is currently on chemotherapy with no adverse reactions above II\u0026deg;.\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eOvarian teratoma was classified into mature teratoma (benign) and immature teratoma (malignant) according to their pathological nature, 95% of which were mature teratoma\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn4\" id=\"#FNLinkFn4\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. Early symptoms are not obvious, mainly to the physical examination found, surgery is the main treatment means currently\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn5\" id=\"#FNLinkFn5\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. SO is a highly specific monoembryonic teratoma, which diagnostic criteria are microscopic thyroid tissue composition\u0026thinsp;\u0026gt;\u0026thinsp;50%, or thyroid tissue\u0026thinsp;\u0026lt;\u0026thinsp;50% but with significant hyperthyroidism, or visually recognizable thyroid tissue in a mature teratoma\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn6\" id=\"#FNLinkFn6\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. SO has predominantly unilateral, left-sided onset, with right-sided onset in slightly older patients and bilateral onset in 8% of patients\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn7\" id=\"#FNLinkFn7\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. The right-sided onset of the disease in this elderly patient is consistent with the literature. The clinical incidence of SO is very low, lacking typical clinical symptoms. Most of them are found by physical examination or with abdominal mass or abdominal pain. However, some studies have reported that about one third of clinical patients have \"Meigs syndrome\"\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn8\" id=\"#FNLinkFn8\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. Based on the high incidence of pleural effusion and ascites and high level of CA125 expression in struma ovarii, some experts suggest that pleural effusion, ascites, and elevated CA125 should be included in the differential diagnosis of pelvic mass and struma ovarii\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn9\" id=\"#FNLinkFn9\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. However, CA125 will increase in both benign and malignant cases, and will not continue to rise even in malignant cases. It is speculated that the increase in CA125 is not a direct result of the existence of the tumor itself, but rather a side effect of ascites\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn10\" id=\"#FNLinkFn10\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. It is worth noting that although SO is a tumor composed of thyroid tissue, only 8% of the patients have hyperthyroidism \u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn11\" id=\"#FNLinkFn11\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. In this case, thyroid function was not tested because the SO was not diagnosed preoperatively, and the patient did not show symptoms of hyperthyroidism. This suggests that thyroid tissue of SO does not have the full function of synthetic thyroid hormone in most cases, but studies show that thyroglobulin can be used as a diagnostic reference for SO\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn12\" id=\"#FNLinkFn12\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e.\u003c/p\u003e \u003cp\u003eBecause of the clinical features of SO are also similar to those of ovarian malignancies, so preoperative imaging diagnosis becomes more important to distinguish ovarian cancer and avoid cancer-type surgery (eg, bilateral salpingectomy, hysterectomy). Ultrasound is the first choice for evaluation of ovarian masses during imaging studies\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn13\" id=\"#FNLinkFn13\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. However, the ultrasound findings of struma ovarii are ambiguous, usually manifests as a multilocular cystic ovarian mass with solid components of various amounts, the ultrasound typically demonstrates these non-specific heterogeneous solid cystic features\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn14\" id=\"#FNLinkFn14\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. The SO has features overlapping with those of malignant ovarian epithelial tumors, though they both present as a unilateral complex adnexal mass often associated with ascites, or as multi-cystic mass with solid components and multiple cystic locules, usually including teratoma components. Familiarity with the \"doughnut sign\" and the \"fat layering sign\" can help in the differential diagnosis, but it is possible to overestimate the malignancy of SO\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn15\" id=\"#FNLinkFn15\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. As SO is an uncommon tumor, unlike the most common types of teratoma, does not demonstrate lipid material on either CT or MRI\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn16\" id=\"#FNLinkFn16\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e, but 123I or 131I scintigraphy is useful for diagnosing a hyper functional SO\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn17\" id=\"#FNLinkFn17\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. Because SO is difficult to make a definitive preoperative diagnosis, postoperative paraffin pathology combined with immunohistochemistry is still the ultimate basis for diagnosis. In addition to thyroid tissue, there was obvious mature teratoma tissue\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn18\" id=\"#FNLinkFn18\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e, typical presentation: multilocular, uneven wall thickness, yellow and grayish-red jelly-like contents; immunohistochemical features: TTF 1 (+), TG (+), PAX-8 (+), CK7 (+), and Muc-1 (+). Due to the low incidence of SO and no clear diagnosis and treatment guidelines, surgery is still the main treatment modality. Due to its ultrasound morphology, which is quite similar to that of malignant ovarian carcinoma, most SO cases are often operated on with laparotomy, involving either ovariotomy or oophorectomy \u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn19\" id=\"#FNLinkFn19\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. For malignant struma ovarii, radioactive iodine supplementation after surgery is effective in preventing metastasis or recurrence\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn20\" id=\"#FNLinkFn20\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e.\u003c/p\u003e \u003cp\u003eIn addition to teratoma and SO of the pathological components, some malignant cells also be found which have significant spindle shape changes, including both epithelial markers and sarcoma markers. At first, we thought it was carcinosarcoma. However, carcinosarcoma needs to determine the components of carcinoma, such as adenocarcinoma, squamous carcinoma. Sarcoma also needs to identify the components, such as leiomyosarcoma, liposarcoma. Which has no clear carcinoma and sarcoma components named sarcomatoid carcinoma (SC)\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn21\" id=\"#FNLinkFn21\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. The specific origin of the cancerous and sarcomatous components could not be determined in this case. The protein P40, which characterizes the most common squamous carcinoma of teratoma malignancy, is negative. But it expressed both epithelial (CK) and mesenchymal component (Vimentin) markers, so pathologists prefer to diagnose sarcomatoid carcinoma. SC is also known as spindle cell carcinoma, where the epithelial carcinoma component determines the biological behavior of metastasis, and the sarcoma component determines the prognosis. Usually, sarcomatoid tissue accounts for more than 50% of malignant tissues\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn22\" id=\"#FNLinkFn22\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. In addition, it is necessary to perform immunohistochemistry to confirm the presence of epithelial components or sarcomatoid tissue surrounded by epithelial tissue that has heavy heterogeneous proliferation of carcinoma in situ. Otherwise, it should be differentiated from sarcoma, so as to avoid misdiagnosis when the proportion of sarcoma components in SC tissue is too large\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn23\" id=\"#FNLinkFn23\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e.\u003c/p\u003e \u003cp\u003eWhether it is carcinosarcoma or sarcomatoid carcinoma, for germ cell tumors, which may have the characteristics of cell stemness, there are currently three theories of its formation mechanism: collision theory, combinatorial theory and transformation theory. \"collision theory\" believes that these two types of diseases have two components: epithelial cancer and sarcomatoid stroma, so it is proposed that tumor cells are biclonal, epithelial cancer components and sarcoma components are derived from two types of stem cells, evolve independently, and then collide; the \"combination theory\" suggests that a stem cell precursor in the early bidirectional differentiation into epithelial carcinoma components and sarcoma components; at present, the most recognized \"transformation theory\" proposes that the sarcoma component comes from carcinoma cells, or the original stem cells is differentiated into one kind of cell and re-differentiated to form a second cell. Its essence is a special type of cancer, and the sarcomatoid component is only the transformation of cancer component\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn24\" id=\"#FNLinkFn24\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. A recent study tested RNA for cancer and sarcoma components in 18 patients with ovarian carcinosarcoma (OCS) found that the cancer component in OCS was more mesenchymal compared to epithelial ovarian cancer, supporting the conjecture that the sarcoma component is transformed through epithelial-mesenchymal cancer component. In this case, CK (+) and Vim (+) suggested that the spindle cell area was characterized by biphasic differentiation characteristics of epithelial and mesenchymal origin, which also tended to support the \"transformation theory\"\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn25\" id=\"#FNLinkFn25\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e.\u003c/p\u003e \u003cp\u003eAlthough sarcomatoid carcinoma has been found in several organs, including the breast, bladder and kidney\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn26\" id=\"#FNLinkFn26\"\u003e\u003c/a\u003e\u003csup\u003e\u0026minus;\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn27\" id=\"#FNLinkFn27\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e, but there are few clinical reports and studies on ovarian sarcomatoid carcinoma (OSC), and only a few documents on PubMed, and most of them are case reports\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn28\" id=\"#FNLinkFn28\"\u003e\u003c/a\u003e\u003csup\u003e,\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn29\" id=\"#FNLinkFn29\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e.There are no standard guidelines for their diagnosis and treatment currently. Combined with the existing literature and our clinical experience, the early symptoms of OSC are not obvious, and they are usually found in physical examination or abdominal pain, abdominal distension and other manifestations. Tumor markers generally increase in CA125, and then the pelvic mass is found by imaging examination. Ultrasound cannot differentiate ovarian sarcomatoid carcinoma from other poorly differentiated ovarian cancers. CT can provide the condition around the lesion and the presence of peritoneal and lymph node metastasis, providing an important basis for clinical staging. MRI shows mixed image of hemorrhage, cyst or necrosis \u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn30\" id=\"#FNLinkFn30\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. Due to the difficulty of preoperative diagnosis of OSC, it is finally determined by postoperative pathological examination, as a result the condition has often progressed to advanced stages when be identified. There is no direct relevant literature for evaluation of prognosis, which can refer to the latest literature report of ovarian carcinosarcoma, which 5-year survival rate is 29.8%, and the median survival period is 16 to 24 months \u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn31\" id=\"#FNLinkFn31\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e.\u003c/p\u003e \u003cp\u003eCurrently, the treatment of OSC is based on cytoreductive surgery combined with platinum-based chemotherapy, similar to ovarian carcinosarcoma or epithelial carcinoma\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn32\" id=\"#FNLinkFn32\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. Among other treatments, radiotherapy is not recommended, and the efficacy of targeted therapy and immune checkpoint inhibitors for OSC is still controversial and needs more studies\u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn33\" id=\"#FNLinkFn33\"\u003e\u003c/a\u003e\u003csup\u003e,\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn34\" id=\"#FNLinkFn34\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. Aoki M et al found that the loss of expression of the ARID1A gene and the same PIK3CA mutation in different tissues, proving the monoclonality of sarcomatoid carcinoma and also providing evidence to support the transformation theory \u003csup\u003e[\u003c/sup\u003e\u003ca class=\"FNLink\" href=\"#Fn35\" id=\"#FNLinkFn35\"\u003e\u003c/a\u003e\u003csup\u003e]\u003c/sup\u003e. According to the monoclonality of the disease, individual treatment options can be selected for the mutation gene to improve the survival rate and improve the quality of life for patients.\u003c/p\u003e \u003cp\u003e \u003cstrong\u003eLimitations\u003c/strong\u003e \u003cp\u003e(1) Although the patient was initially diagnosed as teratoma by color ultrasound and MRI, the patient had no history of thyroid disease or hyperthyroidism symptoms. Therefore, the relevant test was not carried out, and the struma ovarii was not diagnosed before surgery; (2) Up to now, the observation time is short, and the final survival and outcome still need to wait; (3) Genetic testing should be helpful to the diagnosis and treatment of this disease, but because its consumption is not covered by the Chinese medical insurance, so not be given to patients. In the future, we will apply for funding for genetic testing to find more clues about the pathogenesis from the genetic level.\u003c/p\u003e \u003c/p\u003e "},{"header":"Abbreviations","content":"\u003cp\u003e\u003cstrong\u003eSO \u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp; Struma Ovarii\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eMRI \u0026nbsp; \u0026nbsp; \u0026nbsp; Magnetic resonance imaging\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eVD \u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp;Vaginal delivery\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eSC \u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp; Sarcomatoid carcinoma\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eOCS \u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp;Ovarian carcinosarcoma\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eOSC \u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp;Ovarian sarcomatoid carcinoma\u003c/strong\u003e\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eEthics approval and consent to participate\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eNot applicable.\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent for publication\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eThis study obtained informed consent from the patient.\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAvailability of data and materials\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eNot applicable.\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCompeting interests\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eThe authors declare no conflict of interest.\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFunding\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eNot applicable.\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAuthors\u0026apos; contributions\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eH wrote the main manuscript text and was a surgical participant; D and S are surgical participants; J Managed the patient; B provide pathological diagnosis and specimen pictures; Y revised manuscript, the main participant in the surgery.\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAcknowledgments\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eNot applicable.\u003c/strong\u003e\u003c/p\u003e\n"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eGadducci A, Guerrieri ME, Cosio S. 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Insights Imaging.2014 Feb;5(1):41-51.\u003c/li\u003e\n\u003cli\u003eDardik RB, Dardik M ,Westra W, et al. Malignant struma ovarii; two case reports and review of the literature. Gynecol Oncol 1999;73:447-51.\u003c/li\u003e\n\u003cli\u003eOikonomou C, Spathari N, Doumoulaki S, et al. Recurrent Struma Ovarii Presented with High Levels of Thyroglobulin. Case Rep Surg. 2021 Mar 22;2021:8868095.\u003c/li\u003e\n\u003cli\u003eTimmerman D, Planchamp F, Bourne T, et al. ESGO/ISUOG/IOTA/ESGE Consensus Statement on pre-operative diagnosis of ovarian tumors. Int J Gynecol Cancer.2021 Jul;31(7):961-982.\u003c/li\u003e\n\u003cli\u003eOutwater EK, Siegelman ES, Hunt JL(2001)Ovarian teratomas: tumor types and imaging characteristics. Radiographics 21:475\u0026ndash;490 \u003c/li\u003e\n\u003cli\u003eStrachowski LM, Jha P, Phillips CH, et al. O-RADS US v2022: An Update from the American College of Radiology\u0026apos;s Ovarian-Adnexal Reporting and Data System US Committee. 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Sarcomatoid Carcinoma of the Ascending Colon: A Case Report and Literature Review. Am J Case Rep. 2022 Sep 30;23:e937548.\u003c/li\u003e\n\u003cli\u003eHo GY, Kyran EL, Bedo J, et al. Epithelial-to-Mesenchymal Transition Supports Ovarian Carcinosarcoma Tumorigenesis and Confers Sensitivity to Microtubule Targeting with Eribulin. Cancer Res. 2022 Dec 2;82(23):4457-4473.\u003c/li\u003e\n\u003cli\u003eBhargava A, Agrawal S. Sarcomatoid Carcinoma of the Breast: An Unusual Clinical Presentation. Cureus. 2024 Jan 21;16(1):e52696.\u003c/li\u003e\n\u003cli\u003eBrunelli M, Gobbo S, Malpeli G, et al. TROP-2, NECTIN-4 and predictive biomarkers in sarcomatoid and rhabdoid bladder urothelial carcinoma. Pathologica. 2024 Feb;116(1):55-61.\u003c/li\u003e\n\u003cli\u003eGama A, Xu H, Yang XJ, Choy B. Chromophobe Renal Cell Carcinoma with Sarcomatoid Differentiation: Clinicopathologic Correlation and Molecular Findings. Int J Surg Pathol. 2024 Feb;32(1):11-16.\u003c/li\u003e\n\u003cli\u003eUchime KE, Akinjo OA, Awolola NA, et al. A mural nodule of anaplastic carcinoma with sarcomatoid differentiation in a background of ovarian borderline mucinous cystadenoma. Ecancer medical science. 2023 Jun 5;17:1557.\u003c/li\u003e\n\u003cli\u003eHaight P, Savage J, Bixel K. The poor prognosis of sarcomatoid carcinoma arising from low grade serous ovarian cancer: A case report and review of the literature. Gynecol Oncol Rep. 2021 Feb 23;36:100735.\u003c/li\u003e\n\u003cli\u003eSaida T, Mori K, Tanaka YO, et al. Carcinosarcoma of the ovary: MR and clinical findings compared with high-grade serous carcinoma. Jpn J Radiol. 2021 Apr;39(4):357-366.\u003c/li\u003e\n\u003cli\u003eIsmail A, Choi S, Boussios S. Frontiers of Ovarian Carcinosarcoma. Curr Treat Options Oncol. 2023 Dec;24(12):1667-1682.\u003c/li\u003e\n\u003cli\u003eArmstrong DK, Alvarez RD, Backes FJ, et al. NCCN Guidelines\u0026reg; Insights: Ovarian Cancer, Version 3.2022. J Natl Compr Canc Netw. 2022 Sep;20(9):972-980.\u003c/li\u003e\n\u003cli\u003eGarg G, Shah JP, Kumar S, et al. Ovarian and uterine carcinosarcomas: a comparative analysis of prognostic variables and survival outcomes. Int J Gynecol Cancer. 2010 Jul;20(5):888-94.\u003c/li\u003e\n\u003cli\u003eDaniyal M, Polani AS, Canary M. Ovarian Carcinosarcoma and Response to Immunotherapy. Cureus. 2023 Apr 5;15(4):e37149.\u003c/li\u003e\n\u003cli\u003eAoki M, Takaya H, Otani T, et al. Ovarian teratoid carcinosarcoma with a PIK3CA mutation: a case report and review of the literature. Int Cancer Conf J. 2022 Aug 8;11(4):231-237.\u003c/li\u003e\n\u003c/ol\u003e\n"}],"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":"bmc-womens-health","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"bmwh","sideBox":"Learn more about [BMC Women's Health](http://bmcwomenshealth.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/bmwh/default.aspx","title":"BMC Women's Health","twitterHandle":"","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"teratoma, struma ovarii, ovarian sarcomatoid carcinoma, transformation theory","lastPublishedDoi":"10.21203/rs.3.rs-4453757/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-4453757/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003eThis is a rare case of struma ovarii combined with sarcomatoid carcinoma. Whether it's struma ovarii or ovarian sarcomatoid carcinoma, the incidence of both is extremely low and rarely reported, so there are no clear guidelines for either disease and to our knowledge this may be the first case of combined occurrence of both. Therefore, this report describes its clinical manifestations, diagnosis and treatment, analyzes the pathogenesis, and summarizes the previous literature in the hope that it can be helpful to other tumor-related medical personnel and provide material support for the formation of guidelines in this disease.\u003c/p\u003e","manuscriptTitle":"Teratoma combined with struma ovarii and sarcomatoid carcinoma：A case report and review of the literature","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-06-06 18:44:40","doi":"10.21203/rs.3.rs-4453757/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Revision 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