{"paper_id":"63bcfdba-a447-445d-a275-ebf2a4a7d5a1","body_text":"Review began\n 11/05/2024 \nReview ended\n 11/05/2024 \nPublished\n 11/07/2024\n© Copyright \n2024\nThanasa et al. This is an open access\narticle distributed under the terms of the\nCreative Commons Attribution License CC-\nBY 4.0., which permits unrestricted use,\ndistribution, and reproduction in any\nmedium, provided the original author and\nsource are credited.\nDOI:\n 10.7759/cureus.73258\nA Rare Occurrence of a Huge Ovarian Thecoma in\na Menopausal Patient, 25 Years After Abdominal\nHysterectomy and Right Salpingo-Oophorectomy:\nA Case Report and Mini-Review of the Literature\nAnna Thanasa \n, \nEfthymia Thanasa \n, \nIoannis-Rafail Antoniou \n, \nGerasimos Kontogeorgis \n,\nEmmanouil M. Xydias \n, \nApostolos C. Ziogas \n, \nIoannis Thanasas \n1.\n Department of Health Sciences, Medical School, Aristotle University of Thessaloniki, Thessaloniki, GRC \n2.\nDepartment of Obstetrics and Gynecology, General Hospital of Trikala, Trikala, GRC \n3.\n Department of Obstetrics and\nGynecology, EmbryoClinic IVF, Thessaloniki, GRC \n4.\n Department of Obstetrics and Gynecology, University of Thessaly,\nLarissa, GRC\nCorresponding author: \nIoannis Thanasas, \nthanasasg@hotmail.com\nAbstract\nOur case involves a 68-year-old postmenopausal patient with a history of total abdominal hysterectomy and\nright salpingo-oophorectomy performed 25 years ago. The patient presented with chronic pelvic pain for a\ngynecological examination. Clinically, a large painless pelvic mass was palpable, likely originating from the\npreserved left ovary. Transvaginal ultrasonography, computed tomography, and magnetic resonance\nimaging supported the clinical suspicion, and an exploratory laparotomy was subsequently performed.\nIntraoperatively, a solid, neoplastic, exophytic ovarian tumor was discovered, occupying the entire pelvis.\nThe tumor, along with the ovary and the corresponding fallopian tube, was surgically removed. Histological\nexamination of the specimen confirmed the diagnosis of an ovarian thecoma. Following a four-day\nhospitalization with an uneventful postoperative course, the patient was discharged from our clinic. Three\nmonths later, she reported complete relief from chronic pelvic pain. This report of the case also includes a\nbrief literature review, emphasizing the preoperative diagnostic challenges of this rare clinical entity and the\nimportance of regular gynecological check-ups for women who have undergone total hysterectomy with\novarian preservation.\nCategories:\n Obstetrics/Gynecology, Radiology, Oncology\nKeywords:\n case report, chronic pelvic pain, computed tomography, magnetic resonance imaging, ovarian thecoma,\nsurgical treatment, transvaginal ultrasound\nIntroduction\nChronic pelvic pain in women is defined as persistent, non-menstrual pain located in the pelvis, lasting for\nmore than six months \n[1]\n. It is a significant social issue, estimated to affect up to 20% of women \n[2]\n. Chronic\npelvic pain accounts for approximately 40% of laparoscopies and 12% of hysterectomies performed annually\nin the United States, despite the fact that in 80% of cases, its etiology is not gynecologic \n[3]\n. The\netiopathogenesis of chronic pelvic pain is typically non-specific, as it is a multifactorial disorder. It is often\nlinked to dysfunction of the female reproductive system, including conditions such as endometriosis,\nadenomyosis, uterine leiomyomas, ovarian cystic neoplasms, thecomas, fibromas, and ovarian\nfibrothecomas. Less commonly, it may be associated with disorders of the urinary, gastrointestinal, or pelvic\nnervous systems, all of which can significantly impact the quality of life \n[4,5]\n.\nOvarian thecomas, fibromas, and fibrothecomas are rare, non-cancerous sex cord/stromal tumors \n[6]\n. Sex\ncord/stromal tumors account for less than 5% of all ovarian neoplasms \n[7]\n. Thecomas are rare benign tumors\nof the ovarian stroma that can secrete estrogen, androgens, or a combination of both. They are estimated to\nrepresent less than 1% of all benign ovarian neoplasms and typically occur in premenopausal and\npostmenopausal women aged 50-60 years \n[8]\n. The occurrence of thecomas in childhood and adolescence is\nexceedingly rare \n[9]\n. Ovarian thecomas are usually unilateral, with tumor sizes ranging from 5 cm to 10 cm,\nand bilateral involvement in only 3% of cases \n[10]\n. In extremely rare instances, ovarian thecomas have been\nreported during pregnancy \n[7]\n.\nThis paper presents the case of a 68-year-old woman diagnosed with a large ovarian thecoma, 25 years after\nundergoing a total abdominal hysterectomy with unilateral salpingo-oophorectomy. The case underscores\nthe preoperative diagnostic challenges associated with this rare ovarian neoplasm. Additionally, it highlights\nthe importance of regular pelvic examinations in women who have undergone total hysterectomy with\novarian preservation, irrespective of the presence or absence of pelvic symptoms such as chronic pelvic pain.\nCase Presentation\n1\n1\n2\n2\n3\n4\n2\n \nOpen Access Case Report\nHow to cite this article\nThanasa A, Thanasa E, Antoniou I, et al. (November 07, 2024) A Rare Occurrence of a Huge Ovarian Thecoma in a Menopausal Patient, 25 Years\nAfter Abdominal Hysterectomy and Right Salpingo-Oophorectomy: A Case Report and Mini-Review of the Literature. Cureus 16(11): e73258. \nDOI\n10.7759/cureus.73258\n\nA 68-year-old patient, with a history of two vaginal deliveries and an abdominal total hysterectomy with\nright salpingo-oophorectomy performed 25 years ago due to uterine leiomyomas and menometrorrhagia,\nwas referred from the gastroenterology department to the gynecology department for further investigation of\nchronic pelvic pain. The onset of the pain dated back approximately 18 months and was described as mild,\ndeep, constant, and occurring daily. A colonoscopy revealed no abnormal findings and clinical and imaging\nexaminations of the urinary tract showed no lesions affecting the kidneys, ureters, or bladder. The patient's\nmedical history included well-controlled arterial hypertension and hypothyroidism. Her family history was\nunremarkable.\nDuring the vaginal examination, a large, painless pelvic mass was detected, occupying the pouch of Douglas.\nTransvaginal ultrasound revealed a well-defined echogenic mass with both solid and cystic components,\nfilling the entire pelvis (Figure \n1\n).\nFIGURE\n 1: Transvaginal ultrasound imaging of the ovarian thecoma\nA well-defined pelvic mass (yellow arrows) is visible, primarily solid with some cystic elements (red arrows)\nA computed tomography (CT) scan confirmed a large lesion with cystic and solid elements, showing\nheterogeneous enhancement, which appeared to arise from the left ovary. A small collection of free fluid\nwas noted in the Douglas space (Figure \n2\n).\n \n2024 Thanasa et al. Cureus 16(11): e73258. DOI 10.7759/cureus.73258\n2\n of \n8\n\nFIGURE\n 2: CT imaging of the ovarian thecoma\nA large mass with heterogeneous enhancement is evident (yellow arrows), originating from the left ovary and\noccupying the Douglas space\nMagnetic resonance imaging (MRI) demonstrated a lesion with low signal intensity on the T1 sequence. On\nthe T2 sequence, the lesion showed low signal intensity and signs of cystic degeneration. It appeared to be\nhomogeneously enhanced, with no evidence of abnormal lymph nodes in the surrounding area (Figure \n3\n).\nFIGURE\n 3: MRI of the ovarian thecoma\nThe lesion shows low-intensity signal in both T1 and T2 sequences (yellow arrows) with signs of cystic\ndegeneration (red arrows)\nLaboratory tests (Table \n1\n) were conducted to complete the preoperative assessment, and surgical\nmanagement by laparotomy was decided.\n \n2024 Thanasa et al. Cureus 16(11): e73258. DOI 10.7759/cureus.73258\n3\n of \n8\n\nLaboratory Tests\nPreoperative Values\nLaboratory Reference Values\nHt\n41%\n37.7 – 49.7%\nHb\n13.1 gr/dl\n11.8 – 17.8 gr/dl\nWBCc\n7.1x10\n3\n/ml\n4 – 10.8 x10\n3\n/ml\nNEUT\n63%\n40 – 75%\nGlu\n107 mg/dl\n75 – 115 mg/dl\nU\n51 mg/dl\n10 – 50 mg/dl\nCr\n0.9 mg/dl\n0.40 – 1.10 mg/dl\nCEA\n3.45 ng/mL\n< 5 ng/mL\nCA 125\n17.8 U/mL\n≤ 35 U/mL\nCA 15-3\n14.1 U/mL\n0.0 – 31.3 U/mL\nCA 19-9\n13.4 U/mL\n0.0 – 37 U/mL\nTABLE\n 1: Laboratory tests as part of the patient's preoperative check-up\nHt: hematocrit; Hb: hemoglobin; WBC: white blood cells; NEUT: neutral; Glu: glucose; U: urea; Cr: creatinine; CEA: carcinoembryonic antigen; CA: cancer\nantigen\nIntraoperatively, a large ovarian mass, predominantly solid with some cystic elements, was identified,\nmeasuring approximately 15 cm in diameter. The mass was wedged in the pelvis but showed no infiltration\nof adjacent tissues (Figure \n4\n).\nFIGURE\n 4: Intraoperative imaging of the ovarian thecoma\nA solid ovarian mass is observed, emerging from the pelvis without adhering to adjacent tissues\nAfter ligation of the left infundibulopelvic ligament, the neoplastic ovarian tumor was excised along with\nthe left ovary and fallopian tube (Figure \n5\n).\n \n2024 Thanasa et al. Cureus 16(11): e73258. DOI 10.7759/cureus.73258\n4\n of \n8\n\nFIGURE\n 5: Operative specimen of the ovarian thecoma\nA large, oval, exophytic ovarian mass (ovarian thecoma) is clearly visible\nHistological examination confirmed the diagnosis of an ovarian thecoma. The specimen was an oval mass\nweighing 680 grams with a smooth, whitish outer surface. Cross-sections revealed cysts containing serous\nfluid and solid areas with a bundled appearance, whitish color, fibroelastic texture, and focal cystic\ndegeneration, with no evidence of atypia or mitoses (Figure \n6\n).\nFIGURE\n 6: Histological image of the ovarian thecoma\nDepiction of uniform cells with pale cytoplasm, showing no nuclear atypia and no mitoses (Hematoxylin-Eosin\nstain, x20 magnification)\nImmunohistochemical analysis demonstrated strong positivity for inhibin (+++), calretinin (+++), Wilms'\n \n2024 Thanasa et al. Cureus 16(11): e73258. DOI 10.7759/cureus.73258\n5\n of \n8\n\nTumor Protein (WT1) (+++), and vimentin (+++) (Figure \n7\n).\nFIGURE\n 7: Immunohistochemical images of the ovarian thecoma\n(A) Positive staining for calretinin; (B) Positive staining for inhibin; (C) Positive staining for vimentin; (D) Positive\nstaining for Wilms' tumor protein\nAfter a four-day hospitalization with an uneventful postoperative recovery, the patient was discharged.\nThree months later, she reported complete relief from her chronic pelvic pain.\nDiscussion\nThe etiopathogenetic mechanisms of sex cord/stromal ovarian neoplasms have not yet been fully elucidated.\nSex cord/stromal ovarian tumors are rare ovarian neoplasms that include various tumor subtypes with\ndiverse histological features and biological behaviors \n[11]\n. Ovarian thecomas are classified as a subtype of\nstromal tumors according to the World Health Organization classification, revised in 2014 \n[12]\n. Ovarian\nthecomas consist of stromal cells containing lipids (theca cells), which surround ovarian follicles and\ntypically exhibit estrogenic activity \n[13]\n. Additionally, genetic syndromes such as Peutz-Jeghers syndrome\nand DICER1 syndrome are thought to be associated with the pathogenesis of ovarian thecomas \n[14]\n.\nThe clinical manifestations of ovarian thecomas are non-specific, making preoperative diagnosis challenging\n[15]\n. Most patients with ovarian thecoma are asymptomatic, particularly when the tumors are small in size\n[16]\n. Symptoms such as abdominal distention, vague abdominal discomfort, and chronic pelvic pain, as seen\nin our patient, are typically associated with larger thecomas \n[17]\n. In addition, ovarian thecomas frequently\ncause endocrine disorders due to hormone secretion. The secretion of androgenic hormones can result in\noligomenorrhea and the gradual development of secondary male characteristics in women, such as excessive\nhair growth, facial acne, and clitoral hypertrophy \n[18]\n. Rarely, in postmenopausal women, ovarian thecomas\ncan cause endometrial hypertrophy and vaginal bleeding, making it imperative to differentiate them from\nuterine malignancies \n[19]\n. In very rare, isolated cases, ovarian thecoma may also be associated with Meigs\nsyndrome \n[20]\n. The presence of ascites is typically linked to larger tumors, regardless of cancer antigen 125\n(CA-125) levels in the blood serum \n[21]\n. Additionally, in extremely rare instances, thecomas may occur\nduring pregnancy, with the first clinical manifestation being acute abdominal pain caused by pedicle torsion\n[7]\n. In our patient, the diagnosis of ovarian thecoma was delayed. The patient’s history of hysterectomy\nlikely hindered regular gynecological screenings, resulting in a missed diagnosis during the asymptomatic\nphase. The patient presented for examination at the gynecological clinic when the ovarian thecoma had\nreached a larger size, causing chronic pelvic pain due to increased pelvic pressure.\nThe use of modern imaging modalities plays a crucial role in the preoperative diagnosis of ovarian thecomas.\nTransvaginal ultrasound and Doppler ultrasound imaging of the ovaries are first-line imaging examinations.\nThe typical sonographic features of ovarian thecomas include hypoechoic solid masses with well-defined\nborders and the presence of striated shadows, with minimal flow signals on Doppler ultrasonography\n \n2024 Thanasa et al. Cureus 16(11): e73258. DOI 10.7759/cureus.73258\n6\n of \n8\n\n[16,22]\n. MRI is a highly useful diagnostic tool for the female reproductive system and is thought to\nsignificantly improve the accuracy of preoperative diagnosis of ovarian thecomas \n[23]\n. In 2024, Zheng et al.\nhighlighted the utility of MRI in the differential diagnosis of benign thecomas/fibromas from solid\nmalignant ovarian neoplasms in their study \n[24]\n. Although CT cannot replace the importance of evaluating\novarian tumors with MRI, it can still be a useful tool for diagnosing ovarian thecomas/fibromas. The compact\nconsistency of the tumor, its unilateral location, and the absence of enlarged lymph nodes or peritoneal\nmetastases are characteristic CT findings that aid in the early diagnosis of ovarian thecoma/fibroma. These\nfindings help reduce unnecessary referrals of patients to tertiary oncology centers, alleviating the stress that\ncan arise from misdiagnosis of ovarian malignancy \n[25]\n. In our patient, the imaging results indicated the\nbenign nature of the pelvic mass, which influenced our decision to proceed with laparotomy at our hospital\nrather than referring the patient to a tertiary gynecological oncology center. This approach spared the\npatient unnecessary stress and also helped avoid additional hospitalization costs.\nSurgery remains the cornerstone in the treatment of ovarian thecomas. A conservative approach (tumor\nresection or unilateral salpingo-oophorectomy) is preferred in young women, where fertility preservation for\nfuture pregnancy is a primary goal. In older patients, more radical procedures, such as bilateral salpingo-\noophorectomy with or without hysterectomy, depending on the patient’s overall condition, are generally the\nrecommended treatment options \n[6]\n. In our patient, the only viable treatment was the surgical removal of\nthe thecoma along with the corresponding ovary and fallopian tube. Of course, confirmation of the diagnosis\nof ovarian thecoma requires a histological examination of the surgical specimen. Histologically, thecomas\nare characterized by spindle-shaped, oval, or round cells with varying amounts of collagen and a smaller\nproportion of theca cells \n[26]\n. Additionally, microscopic pathological findings such as hyaline plaques,\nnodular growth, calcification, and keloid-like sclerosis are often observed in these neoplasms. In about 40%\nof cases, features of fibroma are also present. Although degenerative atypia is rarely seen, it is essential to\ndifferentiate ovarian thecomas from other stromal ovarian tumors, such as sclerosing stromal tumors,\nmicrocystic stromal tumors, steroid cell tumors, and adult-type granulosa cell tumors of the ovary \n[26]\n. The\nprognosis of ovarian thecomas is generally favorable \n[27]\n.\nConclusions\nThecomas are rare benign ovarian tumors. The occurrence of a large ovarian thecoma many years after a\nhysterectomy with unilateral salpingo-oophorectomy, as seen in our patient, is considered extremely rare.\nFor patients with preserved ovarian tissue, regular pelvic imaging is essential, as it plays a crucial role in the\nearly detection of such neoplasms. Surgical intervention remains the primary treatment for ovarian\nthecomas, emphasizing the importance of vigilant monitoring in these cases.\nAdditional Information\nAuthor Contributions\nAll authors have reviewed the final version to be published and agreed to be accountable for all aspects of the\nwork.\nConcept and design:\n  \nIoannis Thanasas\nAcquisition, analysis, or interpretation of data:\n  \nIoannis Thanasas, Anna Thanasa, Efthymia Thanasa,\nIoannis-Rafail Antoniou, Gerasimos Kontogeorgis, Emmanouil M. Xydias, Apostolos C. Ziogas\nDrafting of the manuscript:\n  \nIoannis Thanasas, Anna Thanasa, Efthymia Thanasa, Ioannis-Rafail\nAntoniou, Gerasimos Kontogeorgis, Emmanouil M. Xydias, Apostolos C. Ziogas\nCritical review of the manuscript for important intellectual content:\n  \nIoannis Thanasas\nSupervision:\n  \nIoannis Thanasas\nDisclosures\nHuman subjects:\n Consent for treatment and open access publication was obtained or waived by all\nparticipants in this study. \nConflicts of interest:\n In compliance with the ICMJE uniform disclosure form, all\nauthors declare the following: \nPayment/services info:\n All authors have declared that no financial support\nwas received from any organization for the submitted work. \nFinancial relationships:\n All authors have\ndeclared that they have no financial relationships at present or within the previous three years with any\norganizations that might have an interest in the submitted work. \nOther relationships:\n All authors have\ndeclared that there are no other relationships or activities that could appear to have influenced the\nsubmitted work.\nReferences\n1\n. \nSpeer LM, Mushkbar S, Erbele T: \nChronic pelvic pain in women\n. 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Pathol Res Pract. 2024, 256:155236. \n10.1016/j.prp.2024.155236\n27\n. \nBurandt E, Young RH: \nThecoma of the ovary: a report of 70 cases emphasizing aspects of its histopathology\ndifferent from those often portrayed and its differential diagnosis\n. Am J Surg Pathol. 2014, 38:1023-32.\n10.1097/PAS.0000000000000252\n \n2024 Thanasa et al. Cureus 16(11): e73258. DOI 10.7759/cureus.73258\n8\n of \n8","source_license":"CC0","license_restricted":false}