A Sex Cord-Stromal Tumor Originating from the Mesorectum: A Case Report

A Sex Cord-Stromal Tumor Originating from the Mesorectum: A Case Report | 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 Sex Cord-Stromal Tumor Originating from the Mesorectum: A Case Report Penghao Li, Yumeng Zhao, Xiaoyu Zhang, Jinshu Ma, Dayong Ding This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-7409319/v1 This work is licensed under a CC BY 4.0 License Status: Under Review Version 1 posted 12 You are reading this latest preprint version Abstract Background: Ovarian sex cord-stromal tumors are rare neoplasms originating from the sex cords and stromal tissue of the ovary, with extraovarian occurrences being even rarer in clinical settings. In this report, we describe a case of an extraovarian sex cord-stromal tumor located in the rectal mesentery, highlighting the diagnostic challenges and management considerations associated with this rare presentation. Case presentation: A 33-year-old Chinese female presented due to a pelvic mass detected during examination. Color Doppler ultrasound revealed a cystic-solid mass located posterior to the uterus, with unclear borders from adjacent intestinal structures; gastrointestinal endoscopy excluded a digestive tract origin, prompting further pelvic Computed Tomography(CT) and Magnetic Resonance Imaging(MRI) evaluation. Imaging indicated a mass lesion anterior to the sacrum with multilocular septations, necessitating differentiation from mucinous tumors; in addition, changes in the left adnexal region suggested an endometriotic cyst. The patient underwent laparoscopic tumor resection; intraoperatively, the tumor was found tightly adherent to the rectal mesentery and primarily vascularized by mesenteric vessels, and was completely excised. Postoperative histopathology and immunohistochemistry confirmed the diagnosis of a sex cord-stromal tumor. The patient had an uneventful postoperative recovery, with no signs of recurrence observed at the 3-month follow-up. Conclusion: This case highlights the potential for ectopic occurrence of sex cord-stromal tumors, underscoring the need to consider this possibility during clinical evaluation and to distinguish it from other pelvic tumors. Incorporating imaging, intraoperative findings, and pathology is essential for accurate diagnosis and treatment planning. Ovarian sex cord-stromal tumors Mesorectum Immunohistochemistry Precise diagnosis Figures Figure 1 Figure 2 Figure 3 Figure 4 Introduction Ovarian sex cord-stromal tumors (OSCSTs) are a rare class of tumors originating from the ovarian sex cords and stromal tissue, accounting for approximately 7% of all ovarian tumors. Among them, theca cell tumors, a subtype of OSCSTs, are even rarer, comprising only 0.5–1.0% of ovarian tumors according to published data. ( 1 – 4 )Due to their overall low incidence and nonspecific clinical manifestations, coupled with limited awareness among clinicians, OSCSTs have a high rate of preoperative misdiagnosis.( 5 – 8 ) Notably, although most theca cell tumors follow a benign course, some cases present with large pelvic masses exceeding 10 cm in diameter, accompanied by ascites/pleural effusion and markedly elevated serum Cancer Antigen 125 levels, mimicking epithelial ovarian cancer and leading to frequent misdiagnosis.( 3 , 4 , 9 , 10 ) Here, we present a case of an extraovarian sex cord-stromal tumor originating from the rectal mesentery, aiming to provide insight into the mechanisms of ectopic occurrence and differential diagnosis. Case Presentation The patient was a 33-year-old woman of reproductive age (G1P1) with no other significant medical history. She presented to our hospital after a pelvic mass was detected during routine physical examination. Transvaginal ultrasound revealed a 6.9 × 7.0 × 3.9 cm anechoic area posterior to the uterus, with thick septations and rich blood flow, poorly demarcated from adjacent bowel loops. An initial diagnosis of a pelvic mass was made. Colonoscopy showed no abnormalities, confirming the mass was not of intraluminal intestinal origin. Enhanced CT and MRI revealed a cystic-solid mass in the presacral space, with a strong suspicion of mucinous tumor, and suggested correlation with clinical findings(Fig. 1 ). Concurrently, changes in the left adnexal region indicated a possible endometriotic cyst. The patient’s CA125 level was mildly elevated at 79.60 U/ml (normal ≤ 47 U/ml), while human epididymis protein 4 (HE4) was 33.10 pmol/L (normal range 0–70 pmol/L), within normal limit. Liquid-based cervical cytology showed no intraepithelial lesions or malignant cells, and Human Papillomavirus Deoxyribonucleic Acid(HPV DNA) testing was negative. The patient underwent laparoscopic surgery with complete excision of the mass and concurrent removal of the left endometriotic cyst. Intraoperatively, the mass was located within the rectal mesentery, with its main feeding vessels arising from the mesentery(Fig. 2 ). The tumor appeared grossly as a nodular mass, measuring 6.5 × 5 × 3 cm, with a partially smooth capsule. The cut surface was solid and grayish-white, with cystic degeneration in the center, containing clear fluid(Fig. 3 ). Immunohistochemistry showed positive staining for CD34, SMA, SDHB, Inhibin α, Bcl-2, and CD10, while CD117, Dog-1, Desmin, S-100, SF-1, FOXL2, WT-1, and CR were all negative(Fig. 4 ). Based on histological morphology and immunohistochemical profile, the diagnosis was consistent with a thecoma, a subtype of sex cord-stromal tumor. The patient experienced good postoperative recovery of gastrointestinal function, and pelvic MRI at 3-month follow-up showed no evidence of recurrence. Discussion The age distribution of theca cell tumors is distinct, with a predominance in perimenopausal and postmenopausal women, while cases in prepubertal and reproductive-age females are rare.( 2 , 4 ) This case is unique in two respects: first, the patient presented preoperatively with elevated serum CA125 and ascites, which aligns with the known biological behavior of thecoma; second, the tumor originated in the rectal mesentery, an unusual anatomical site, resulting in imaging features that differ markedly from typical adnexal masses and complicating preoperative localization and diagnosis. Moreover, the patient initially presented only with an asymptomatic pelvic mass, without the typical symptoms of thecoma such as abdominal pain, menstrual irregularities, or abnormal uterine bleeding. During preoperative differential diagnosis, common rectal tumors such as rectal carcinoma and rectal stromal tumors were excluded using colonoscopy, contrast-enhanced pelvic MRI, and enhanced pelvic CT. Radiologically, rectal stromal tumors typically appear as oval solid masses, with occasional cystic degeneration and necrosis when large. Rectal leiomyomas are more common in middle-aged and elderly women and exhibit heterogeneous enhancement on contrast CT, often with central necrosis and marked peripheral enhancement.( 11 ) These findings did not align with the imaging profile of our case, further highlighting the diagnostic complexity of ectopic sex cord-stromal tumors. The definitive diagnosis in this case was established through surgical excision and pathological biopsy. Among the diagnostic markers, α-inhibin is highly valuable for OSCSTs; in a study by Deavers et al., 85% of 53 OSCST cases were positive for α-inhibin.( 12 ) The standardized treatment for thecoma centers on complete surgical excision, and the choice of surgical approach must consider the patient's age, fertility desires, tumor biology, and pelvic organ involvement. For women of reproductive age, local tumor excision is recommended to preserve fertility, while postmenopausal women or those with uterine/adnexal comorbidities should undergo ipsilateral adnexectomy or even total hysterectomy with bilateral salpingo-oophorectomy.( 4 , 10 , 13 ) In this case, as the patient was a reproductive-age female (G1P1) and the tumor's nature was unclear preoperatively, an adnexectomy was not performed. Although it remains unclear whether thecoma arising from the rectal mesentery is related to adnexal pathology, this case suggests that: ( 1 ) For cystic-solid pelvic masses in atypical locations, sex cord-stromal tumors should be considered in the differential diagnosis even if imaging features differ from typical adnexal tumors; ( 2 ) Intraoperative frozen section analysis should be routinely performed to enhance diagnostic specificity. Timely recognition of atypical presentations is essential to avoid diagnostic delay or mismanagement. When encountering similar cases in the future, clinicians should remain vigilant for potential diagnostic challenges to avoid missing rare tumors due to conventional diagnostic thinking or delivering unnecessary treatment that could impair quality of life. Conclusion This case achieved a favorable prognosis through radical surgery, with complete preservation of the patient’s reproductive function (regular menstrual cycles postoperatively). However, further investigation is needed into the pathogenesis and clinical features of extraovarian thecomas. Such studies will provide evidence-based support for optimizing diagnostic and therapeutic strategies. This case serves as a reminder to clinicians regarding the differential diagnosis of pelvic masses in atypical anatomical locations. Abbreviations The following abbreviations are used in this manuscript: OSCSTs Ovarian sex cord-stromal tumors CT Computed Tomography MRI Magnetic Resonance Imaging CA125 Cancer Antigen 125 HE4 Human Epididymis protein 4 HPV DNA Human Papillomavirus Deoxyribonucleic Acid Declarations Ethics approval and consent to participate Not applicable Consent for publication Written informed consent for publication of their clinhical details and/or clinical images was obtained from the patient/parent/guardian/ relative of the patint. A copy of the consent form is available for review by the Editor of this journal Availability of data and materials Not applicable Competing interests The authors declare that they have no competing interests Funding Not applicable Authors' contributions P.L. completed the drafting of the main manuscript. D.D. and X.Z. provided the case information and performed the surgery. Y.Z. completed the language review and figure processing for the article. J.M. provided the pathological diagnosis and was responsible for Figure 4. All authors reviewed the manuscript. Acknowledgements Not applicable References Horta M, Cunha TM. Sex cord-stromal tumors of the ovary: a comprehensive review and update for radiologists. Diagnostic and interventional radiology (Ankara, Turkey). 2015;21(4):277-86. Chen VW, Ruiz B, Killeen JL, Coté TR, Wu XC, Correa CN. Pathology and classification of ovarian tumors. Cancer. 2003;97(10 Suppl):2631-42. Nocito AL, Sarancone S, Bacchi C, Tellez T. Ovarian thecoma: clinicopathological analysis of 50 cases. Annals of diagnostic pathology. 2008;12(1):12-6. Wu LY, Li HJ,LL,Kong WM,Liu LY. Clinical analysis of 74 cases with ovarian thecoma. Chinese Journal of Obstetrics and Gynecology.2002(02):40-2. Stuart GC, Dawson LM. Update on granulosa cell tumours of the ovary. Current opinion in obstetrics & gynecology. 2003;15(1):33-7. Tamai K, Koyama T, Saga T, Kido A, Kataoka M, Umeoka S, et al. MR features of physiologic and benign conditions of the ovary. European radiology. 2006;16(12):2700-11. Omori M, Kondo T, Fukushima J, Oi M, Watanabe Y, Nakazawa T, et al. Extraovarian Fibroma With Minor Sex Cord Elements: A Case Report and Literature Review. International journal of surgical pathology. 2017;25(5):472-6. Li Q, Yuan XJ, Kong JZ, Ma XL. MRI Diagnosis and Differential Diagnosis of Ovarian Sex Cord-Stromal Tumors. Chinese Imaging Journal of Integrated Traditional and Western Medicine. 2017;15(01):92-4. Tanaka YO, Tsunoda H, Kitagawa Y, Ueno T, Yoshikawa H, Saida Y. Functioning ovarian tumors: direct and indirect findings at MR imaging. Radiographics : a review publication of the Radiological Society of North America, Inc. 2004;24 Suppl 1:S147-66. Wang XL, Li TF, Li X, et al. CliniCal analysis of 37 Cases wifh Ovarian thecoma. Journal of Modern Oncology. 2008(03):422-4. Yuan F, Ling LF, Lan X, et al. Imaging Manifestations Of Rectal Gastrointestinal Stromal Tumors. Chinese Imaging Journal of Integrated Traditional and Western Medicine. 2024;22(06):729-31. Deavers MT, Malpica A, Liu J, Broaddus R, Silva EG. Ovarian sex cord-stromal tumors: an immunohistochemical study including a comparison of calretinin and inhibin. Modern pathology : an official journal of the United States and Canadian Academy of Pathology, Inc. 2003;16(6):584-90. Ayhan A, Salman MC, Velipasaoglu M, Sakinci M, Yuce K. Prognostic factors in adult granulosa cell tumors of the ovary: a retrospective analysis of 80 cases. Journal of gynecologic oncology. 2009;20(3):158-63. Additional Declarations No competing interests reported. Cite Share Download PDF Status: Under Review Version 1 posted Editorial decision: Revision requested 16 Oct, 2025 Reviews received at journal 02 Oct, 2025 Reviews received at journal 25 Sep, 2025 Reviewers agreed at journal 24 Sep, 2025 Reviewers agreed at journal 24 Sep, 2025 Reviews received at journal 23 Sep, 2025 Reviewers agreed at journal 23 Sep, 2025 Reviewers invited by journal 16 Sep, 2025 Editor invited by journal 22 Aug, 2025 Editor assigned by journal 19 Aug, 2025 Submission checks completed at journal 19 Aug, 2025 First submitted to journal 19 Aug, 2025 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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polyps.\u003c/strong\u003e\u003c/p\u003e","description":"","filename":"1.png","url":"https://assets-eu.researchsquare.com/files/rs-7409319/v1/3aaca109c4189999ce6c453a.png"},{"id":92158018,"identity":"9722d5fb-80a4-46e2-a4be-12ae0fa9cefb","added_by":"auto","created_at":"2025-09-25 09:26:54","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":610403,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cstrong\u003eIntraoperative images Intraoperatively, the tumor was found within the mesorectum and was unrelated to the uterus and ovaries.After dissection of the rectosacral ligament, part of the tumor became visible.The tumor appeared cystic-solid in nature, with feeding vessels originating from the mesorectum.\u003c/strong\u003e\u003c/p\u003e","description":"","filename":"2.png","url":"https://assets-eu.researchsquare.com/files/rs-7409319/v1/ff7957b934964605966b69e6.png"},{"id":92158147,"identity":"8e20923a-b255-412a-a9d6-bbec379348b5","added_by":"auto","created_at":"2025-09-25 09:34:54","extension":"png","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":968103,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cstrong\u003eMacroscopic tumor images A. The tumor was entirely removed, with dimensions comparable to those seen on preoperative scans.\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eB. The tumor was cut open after fixation in formalin, revealing a heterogeneous composition of cystic and solid components.\u003c/strong\u003e\u003c/p\u003e","description":"","filename":"3.png","url":"https://assets-eu.researchsquare.com/files/rs-7409319/v1/9400b1544aab47c9a71041c8.png"},{"id":92156943,"identity":"0f039710-a340-482d-b2db-1fe2144ed9bc","added_by":"auto","created_at":"2025-09-25 09:18:54","extension":"png","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":1576471,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cstrong\u003eHistopathological images of the tumor\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eA and B: Hematoxylin and eosin (H\u0026amp;E) stained sections of the tumor under 20× and 100× magnification.\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eC and D: Immunohistochemistry of the tumor showing (C) BCL-2 and (D) inhibin-α expression.\u003c/strong\u003e\u003c/p\u003e","description":"","filename":"4.png","url":"https://assets-eu.researchsquare.com/files/rs-7409319/v1/94848f05525203e5ed4c7d26.png"},{"id":92159182,"identity":"7d848d0b-d6b1-49c1-a318-cc0abb9a7669","added_by":"auto","created_at":"2025-09-25 09:42:56","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":5742560,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-7409319/v1/9416e02c-bad8-4002-991e-f2325e5d9767.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"\u003cp\u003eA Sex Cord-Stromal Tumor Originating from the Mesorectum: A Case Report\u003c/p\u003e","fulltext":[{"header":"Introduction","content":"\u003cp\u003eOvarian sex cord-stromal tumors (OSCSTs) are a rare class of tumors originating from the ovarian sex cords and stromal tissue, accounting for approximately 7% of all ovarian tumors. Among them, theca cell tumors, a subtype of OSCSTs, are even rarer, comprising only 0.5\u0026ndash;1.0% of ovarian tumors according to published data. (\u003cspan additionalcitationids=\"CR2 CR3\" citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e)Due to their overall low incidence and nonspecific clinical manifestations, coupled with limited awareness among clinicians, OSCSTs have a high rate of preoperative misdiagnosis.(\u003cspan additionalcitationids=\"CR6 CR7\" citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e)\u003c/p\u003e\u003cp\u003eNotably, although most theca cell tumors follow a benign course, some cases present with large pelvic masses exceeding 10 cm in diameter, accompanied by ascites/pleural effusion and markedly elevated serum Cancer Antigen 125 levels, mimicking epithelial ovarian cancer and leading to frequent misdiagnosis.(\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e, \u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e, \u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e, \u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e)\u003c/p\u003e\u003cp\u003eHere, we present a case of an extraovarian sex cord-stromal tumor originating from the rectal mesentery, aiming to provide insight into the mechanisms of ectopic occurrence and differential diagnosis.\u003c/p\u003e"},{"header":"Case Presentation","content":"\u003cp\u003eThe patient was a 33-year-old woman of reproductive age (G1P1) with no other significant medical history. She presented to our hospital after a pelvic mass was detected during routine physical examination. Transvaginal ultrasound revealed a 6.9 \u0026times; 7.0 \u0026times; 3.9 cm anechoic area posterior to the uterus, with thick septations and rich blood flow, poorly demarcated from adjacent bowel loops. An initial diagnosis of a pelvic mass was made.\u003c/p\u003e\u003cp\u003eColonoscopy showed no abnormalities, confirming the mass was not of intraluminal intestinal origin. Enhanced CT and MRI revealed a cystic-solid mass in the presacral space, with a strong suspicion of mucinous tumor, and suggested correlation with clinical findings(Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003e). Concurrently, changes in the left adnexal region indicated a possible endometriotic cyst.\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003eThe patient\u0026rsquo;s CA125 level was mildly elevated at 79.60 U/ml (normal\u0026thinsp;\u0026le;\u0026thinsp;47 U/ml), while human epididymis protein 4 (HE4) was 33.10 pmol/L (normal range 0\u0026ndash;70 pmol/L), within normal limit. Liquid-based cervical cytology showed no intraepithelial lesions or malignant cells, and Human Papillomavirus Deoxyribonucleic Acid(HPV DNA) testing was negative.\u003c/p\u003e\u003cp\u003eThe patient underwent laparoscopic surgery with complete excision of the mass and concurrent removal of the left endometriotic cyst. Intraoperatively, the mass was located within the rectal mesentery, with its main feeding vessels arising from the mesentery(Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003e).\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003eThe tumor appeared grossly as a nodular mass, measuring 6.5 \u0026times; 5 \u0026times; 3 cm, with a partially smooth capsule. The cut surface was solid and grayish-white, with cystic degeneration in the center, containing clear fluid(Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003e).\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003eImmunohistochemistry showed positive staining for CD34, SMA, SDHB, Inhibin α, Bcl-2, and CD10, while CD117, Dog-1, Desmin, S-100, SF-1, FOXL2, WT-1, and CR were all negative(Fig.\u0026nbsp;\u003cspan refid=\"Fig4\" class=\"InternalRef\"\u003e4\u003c/span\u003e).\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003eBased on histological morphology and immunohistochemical profile, the diagnosis was consistent with a thecoma, a subtype of sex cord-stromal tumor. The patient experienced good postoperative recovery of gastrointestinal function, and pelvic MRI at 3-month follow-up showed no evidence of recurrence.\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eThe age distribution of theca cell tumors is distinct, with a predominance in perimenopausal and postmenopausal women, while cases in prepubertal and reproductive-age females are rare.(\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e, \u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e) This case is unique in two respects: first, the patient presented preoperatively with elevated serum CA125 and ascites, which aligns with the known biological behavior of thecoma; second, the tumor originated in the rectal mesentery, an unusual anatomical site, resulting in imaging features that differ markedly from typical adnexal masses and complicating preoperative localization and diagnosis.\u003c/p\u003e\u003cp\u003eMoreover, the patient initially presented only with an asymptomatic pelvic mass, without the typical symptoms of thecoma such as abdominal pain, menstrual irregularities, or abnormal uterine bleeding.\u003c/p\u003e\u003cp\u003eDuring preoperative differential diagnosis, common rectal tumors such as rectal carcinoma and rectal stromal tumors were excluded using colonoscopy, contrast-enhanced pelvic MRI, and enhanced pelvic CT.\u003c/p\u003e\u003cp\u003eRadiologically, rectal stromal tumors typically appear as oval solid masses, with occasional cystic degeneration and necrosis when large. Rectal leiomyomas are more common in middle-aged and elderly women and exhibit heterogeneous enhancement on contrast CT, often with central necrosis and marked peripheral enhancement.(\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e)\u003c/p\u003e\u003cp\u003eThese findings did not align with the imaging profile of our case, further highlighting the diagnostic complexity of ectopic sex cord-stromal tumors.\u003c/p\u003e\u003cp\u003eThe definitive diagnosis in this case was established through surgical excision and pathological biopsy. Among the diagnostic markers, α-inhibin is highly valuable for OSCSTs; in a study by Deavers et al., 85% of 53 OSCST cases were positive for α-inhibin.(\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e)\u003c/p\u003e\u003cp\u003eThe standardized treatment for thecoma centers on complete surgical excision, and the choice of surgical approach must consider the patient's age, fertility desires, tumor biology, and pelvic organ involvement.\u003c/p\u003e\u003cp\u003eFor women of reproductive age, local tumor excision is recommended to preserve fertility, while postmenopausal women or those with uterine/adnexal comorbidities should undergo ipsilateral adnexectomy or even total hysterectomy with bilateral salpingo-oophorectomy.(\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e, \u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e, \u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e)\u003c/p\u003e\u003cp\u003eIn this case, as the patient was a reproductive-age female (G1P1) and the tumor's nature was unclear preoperatively, an adnexectomy was not performed.\u003c/p\u003e\u003cp\u003eAlthough it remains unclear whether thecoma arising from the rectal mesentery is related to adnexal pathology, this case suggests that:\u003c/p\u003e\u003cp\u003e(\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e) For cystic-solid pelvic masses in atypical locations, sex cord-stromal tumors should be considered in the differential diagnosis even if imaging features differ from typical adnexal tumors;\u003c/p\u003e\u003cp\u003e(\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e) Intraoperative frozen section analysis should be routinely performed to enhance diagnostic specificity.\u003c/p\u003e\u003cp\u003eTimely recognition of atypical presentations is essential to avoid diagnostic delay or mismanagement. When encountering similar cases in the future, clinicians should remain vigilant for potential diagnostic challenges to avoid missing rare tumors due to conventional diagnostic thinking or delivering unnecessary treatment that could impair quality of life.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eThis case achieved a favorable prognosis through radical surgery, with complete preservation of the patient\u0026rsquo;s reproductive function (regular menstrual cycles postoperatively).\u003c/p\u003e\u003cp\u003eHowever, further investigation is needed into the pathogenesis and clinical features of extraovarian thecomas. Such studies will provide evidence-based support for optimizing diagnostic and therapeutic strategies.\u003c/p\u003e\u003cp\u003eThis case serves as a reminder to clinicians regarding the differential diagnosis of pelvic masses in atypical anatomical locations.\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cp\u003e\u003cstrong\u003eThe following abbreviations are used in this manuscript:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eOSCSTs \u0026nbsp; \u0026nbsp; Ovarian sex cord-stromal tumors\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCT \u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp; Computed Tomography\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eMRI \u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp;Magnetic Resonance Imaging\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCA125 \u0026nbsp; \u0026nbsp; \u0026nbsp;Cancer Antigen 125\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eHE4 \u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp;Human Epididymis protein 4\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eHPV DNA \u0026nbsp; Human Papillomavirus Deoxyribonucleic Acid\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\u003eNot applicable\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent for publication\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eWritten informed consent for publication of their clinhical details and/or clinical images was obtained from the patient/parent/guardian/ relative of the patint. A copy of the consent form is available for review by the Editor of this journal\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAvailability of data and materials\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNot applicable\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\n\u003cp\u003e\u003cstrong\u003eFunding\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNot applicable\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAuthors\u0026apos; contributions\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eP.L. completed the drafting of the main manuscript.\u003c/p\u003e\n\u003cp\u003eD.D. and X.Z. provided the case information and performed the surgery.\u003c/p\u003e\n\u003cp\u003eY.Z. completed the language review and figure processing for the article.\u003c/p\u003e\n\u003cp\u003eJ.M. provided the pathological diagnosis and was responsible for Figure 4.\u003c/p\u003e\n\u003cp\u003eAll authors reviewed the manuscript.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAcknowledgements\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNot applicable\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eHorta M, Cunha TM. Sex cord-stromal tumors of the ovary: a comprehensive review and update for radiologists. Diagnostic and interventional radiology (Ankara, Turkey). 2015;21(4):277-86.\u003c/li\u003e\n\u003cli\u003eChen VW, Ruiz B, Killeen JL, Cot\u0026eacute; TR, Wu XC, Correa CN. Pathology and classification of ovarian tumors. Cancer. 2003;97(10 Suppl):2631-42.\u003c/li\u003e\n\u003cli\u003eNocito AL, Sarancone S, Bacchi C, Tellez T. Ovarian thecoma: clinicopathological analysis of 50 cases. Annals of diagnostic pathology. 2008;12(1):12-6.\u003c/li\u003e\n\u003cli\u003eWu LY, Li HJ,LL,Kong WM,Liu LY. Clinical analysis of 74 cases with ovarian thecoma. Chinese Journal of Obstetrics and Gynecology.2002(02):40-2.\u003c/li\u003e\n\u003cli\u003eStuart GC, Dawson LM. Update on granulosa cell tumours of the ovary. Current opinion in obstetrics \u0026amp; gynecology. 2003;15(1):33-7.\u003c/li\u003e\n\u003cli\u003eTamai K, Koyama T, Saga T, Kido A, Kataoka M, Umeoka S, et al. MR features of physiologic and benign conditions of the ovary. European radiology. 2006;16(12):2700-11.\u003c/li\u003e\n\u003cli\u003eOmori M, Kondo T, Fukushima J, Oi M, Watanabe Y, Nakazawa T, et al. Extraovarian Fibroma With Minor Sex Cord Elements: A Case Report and Literature Review. International journal of surgical pathology. 2017;25(5):472-6.\u003c/li\u003e\n\u003cli\u003eLi Q, Yuan XJ, Kong JZ, Ma XL. MRI Diagnosis and Differential Diagnosis of Ovarian Sex Cord-Stromal Tumors. Chinese Imaging Journal of Integrated Traditional and Western Medicine. 2017;15(01):92-4.\u003c/li\u003e\n\u003cli\u003eTanaka YO, Tsunoda H, Kitagawa Y, Ueno T, Yoshikawa H, Saida Y. Functioning ovarian tumors: direct and indirect findings at MR imaging. Radiographics : a review publication of the Radiological Society of North America, Inc. 2004;24 Suppl 1:S147-66.\u003c/li\u003e\n\u003cli\u003eWang XL, Li TF, Li X, et al. CliniCal analysis of 37 Cases wifh Ovarian thecoma. Journal of Modern Oncology. 2008(03):422-4.\u003c/li\u003e\n\u003cli\u003eYuan F, Ling LF, Lan X, et al. Imaging Manifestations Of Rectal Gastrointestinal Stromal Tumors. Chinese Imaging Journal of Integrated Traditional and Western Medicine. 2024;22(06):729-31.\u003c/li\u003e\n\u003cli\u003eDeavers MT, Malpica A, Liu J, Broaddus R, Silva EG. Ovarian sex cord-stromal tumors: an immunohistochemical study including a comparison of calretinin and inhibin. Modern pathology : an official journal of the United States and Canadian Academy of Pathology, Inc. 2003;16(6):584-90.\u003c/li\u003e\n\u003cli\u003eAyhan A, Salman MC, Velipasaoglu M, Sakinci M, Yuce K. Prognostic factors in adult granulosa cell tumors of the ovary: a retrospective analysis of 80 cases. Journal of gynecologic oncology. 2009;20(3):158-63.\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":false,"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":"Ovarian sex cord-stromal tumors, Mesorectum, Immunohistochemistry, Precise diagnosis","lastPublishedDoi":"10.21203/rs.3.rs-7409319/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-7409319/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cstrong\u003eBackground:\u003c/strong\u003e Ovarian sex cord-stromal tumors are rare neoplasms originating from the sex cords and stromal tissue of the ovary, with extraovarian occurrences being even rarer in clinical settings. In this report, we describe a case of an extraovarian sex cord-stromal tumor located in the rectal mesentery, highlighting the diagnostic challenges and management considerations associated with this rare presentation.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCase presentation:\u003c/strong\u003e A 33-year-old Chinese female presented due to a pelvic mass detected during examination. Color Doppler ultrasound revealed a cystic-solid mass located posterior to the uterus, with unclear borders from adjacent intestinal structures; gastrointestinal endoscopy excluded a digestive tract origin, prompting further pelvic Computed Tomography(CT) and Magnetic Resonance Imaging(MRI) evaluation. Imaging indicated a mass lesion anterior to the sacrum with multilocular septations, necessitating differentiation from mucinous tumors; in addition, changes in the left adnexal region suggested an endometriotic cyst.\u003cbr\u003e\nThe patient underwent laparoscopic tumor resection; intraoperatively, the tumor was found tightly adherent to the rectal mesentery and primarily vascularized by mesenteric vessels, and was completely excised. Postoperative histopathology and immunohistochemistry confirmed the diagnosis of a sex cord-stromal tumor. The patient had an uneventful postoperative recovery, with no signs of recurrence observed at the 3-month follow-up.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConclusion: \u003c/strong\u003eThis case highlights the potential for ectopic occurrence of sex cord-stromal tumors, underscoring the need to consider this possibility during clinical evaluation and to distinguish it from other pelvic tumors. Incorporating imaging, intraoperative findings, and pathology is essential for accurate diagnosis and treatment planning.\u003c/p\u003e","manuscriptTitle":"A Sex Cord-Stromal Tumor Originating from the Mesorectum: A Case Report","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2025-09-25 09:18:49","doi":"10.21203/rs.3.rs-7409319/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Revision requested","date":"2025-10-16T08:23:58+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-10-02T10:26:23+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-09-25T16:42:10+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"279593005299777663709244885934870170095","date":"2025-09-25T00:52:20+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"307256789578572543249162105069063062951","date":"2025-09-24T19:23:47+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-09-23T09:47:32+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"214726381474380742709227239249412962225","date":"2025-09-23T09:32:05+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2025-09-16T11:33:23+00:00","index":"","fulltext":""},{"type":"editorInvited","content":"","date":"2025-08-22T10:30:22+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2025-08-20T00:08:07+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2025-08-20T00:06:51+00:00","index":"","fulltext":""},{"type":"submitted","content":"BMC Women's Health","date":"2025-08-19T13:41:32+00:00","index":"","fulltext":""}],"status":"published","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}}],"origin":"","ownerIdentity":"4bb40871-8e53-4a50-aeda-67219338c903","owner":[],"postedDate":"September 25th, 2025","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"under-review","subjectAreas":[],"tags":[],"updatedAt":"2025-11-07T06:53:14+00:00","versionOfRecord":[],"versionCreatedAt":"2025-09-25 09:18:49","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-7409319","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-7409319","identity":"rs-7409319","version":["v1"]},"buildId":"8U1c8b4HqxoKbykW_rLl7","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}