Fibrothecoma of broad ligament with minor sex cord elements: Case report and brief literature review.

RATIONALE: Sex cord-stromal tumors are always found in ovary, but the occurrence of this kind of tumor at extraovarian locations is extremely rare. Up to now, the case concerning fibrothecoma of broad ligament with minor sex cord elements has not been reported, and it is extremely challenging to diagnose before surgery. In this case report, we summarized pathogenesis, clinical features, laboratory finding, imaging studies, pathology, and therapeutic schedule of this tumor, with the aim of raising awareness and attention to this type of disease. PATIENT CONCERNS: A 45-year-old Chinese woman was referred to our department with intermittent lower abdominal pain for about 6 years. On examination, both ultrasonography and computed tomography revealed she had a right adnexal mass. DIAGNOSIS: Based on the results of histology and immunohistochemistry, the final diagnosis was confirmed as fibrothecoma of broad ligament with minor sex cord elements. INTERVENTIONS: This patient underwent laparoscopic unilateral salpingo-oophorectomy with excision of the neoplasm. OUTCOMES: Eleven days post-treatment, the patient complained that the symptoms of abdominal pain was disappeared. There is no evidence of disease recurrence 5 years after laparoscopic surgery according to the consequences of radiologic examination. CONCLUSION: The natural history of this kind of tumor is uncertain. Although main treatment of this neoplasm might be surgical resection and good prognosis can be achieved, we believe that long-time follow-up is extremely important in all patients diagnosed as fibrothecoma of broad ligament with minor sex cord. Laparoscopic unilateral salpingo-oophorectomy with excision of the tumor should be recommended to these patients.

Synchronous uterine serous carcinoma and ovarian sex cord stromal tumor (thecoma)-A rare first case report.

Synchronous endometrial and ovarian carcinoma is a rare instance and it accounts for 50 to 70% of all synchronous female genital tract tumors. However, it is very rare to find synchronous endometrial carcinoma and ovarian sex cord-stromal tumor (thecoma). The present case is a 75-year-old woman with a complaint of post-menopausal vaginal bleeding. Radiologically, the magnetic resonance imaging (MRI) pelvis revealed altered signal intensity mass in the uterus. Frozen section and routine histopathological examination were done on radical hysterectomy. Microscopically, serous carcinoma involving uterine corpus and left Fallopian tube was identified along with the unusual finding of contralateral ovarian sex cord-stromal tumor (thecoma), which was confirmed on immunohistochemical examination. It is a very rare association and is first reported in the present study after a thorough search of the published literature. Their relationship based on a high level of estrogen produced by the hyperactive ovary is controversial as serous carcinomas are less hormone-dependent.

Giant Ovarian Thecoma Associated with Meigs Syndrome: A Striking Case.

Introduction: Meigs syndrome consists of the presence of a benign ovarian tumor, ascites and pleural effusion, and the latter two subdued after surgical excision of the ovarian tumor. Elevated Ca-125 in this context is confusing and is reported only in a handful of cases. A rare but striking case with the above features is presented herein. Case Presentation: A 46-year-old woman with a giant pelvic/abdominal mass originating from her right adnexa, ascites and pleural effusion, with elevated Ca-125 (938 IU/mL) was treated with the presumptive diagnosis of stage IV ovarian cancer. Imaging modalities showed a 22 cm solid adnexal mass and the patient underwent total abdominal hysterectomy and bilateral salpigooophorectomy, omentectomy and drainage of 4L of ascetic fluid. Surprisingly, final histopathology was negative for malignancy, characterizing the primary tumor as ovarian thecoma. Ascites and pleural effusion resolved by the seventh postoperative day, setting the diagnosis of Meigs syndrome. Discussion: Meigs syndrome accounts for 1% of all ovarian tumors, however it should be considered in the differential diagnosis when clinicians come across the classic triad of the syndrome, even when Ca-125 is elevated. These patients have normal life expectancy with meticulous management, while pathophysiology of this condition remains uncertain in various points.

Ovarian thecoma: A very unusual cause of postmenopausal bleeding.

We describe a 67-year-old woman with postmenopausal bleeding having ceased estrogen plus progestogen therapy nine months before. Transvaginal ultrasonography showed endometrial thickening with normal ovarian appearance. Hormonal studies revealed high estradiol and inhibin B levels but normal androgens and adrenal hormones. Magnetic resonance image demonstrated a 13-mm left ovarian tumour. Hysterectomy and bilateral salpingo-oophorectomy were performed, and the pathological study revealed an 8 mm ovarian thecoma. This case illustrates a very unusual cause of postmenopausal bleeding. We suggest a study protocol and discuss the differential diagnosis of this case.

COMPLETE REMISSION OF MEDICALLY TREATED LUTEINIZED THECOMAS WITH SCLEROSING PERITONITIS.

We describe the first French patient treated for sclerosing peritonitis syndrome associated with lutheinized thecomas. At 42 years old, she had respiratory distress with increased abdominal volume. Physical examination revealed ascites, pleural effusions, and two mobile latero-uterine masses. Radiological examinations revealed bilateral ovarian masses of 10 cm. Bilateral adnexectomy was performed by laparotomy. Histological analysis concluded that there were benign luteinized thecomas. Until the 36th postoperative day, the general condition of the patient deteriorated to become critical. A second surgical procedure was attempted revealing sclerosing fibrosis preventing access to the peritoneal cavity. Subsequently, a medical treatment combining parenteral nutrition, high intravenous doses of corticosteroids, antiestrogens, colchicine and sandostatin was administered and effective allowing continuity recovery 15 months later. The clinical outcomes has been favorable at 24 month later.

A case of a 20-kg fibrothecoma.

A case report of a 50-cm diameter and 20-kg mass of benign ovarian tumor. Total abdominal hysterectomy with a bilateral salpingooophorectomy was performed with full patient recovery. Fibrothecomas can remain long asymptomatic and can grow to giant sizes.

Contribution of ultrasound in the study of ovarian fibrothecomas: a series of 47 cases.

INTRODUCTION: Ovarian fibro-thecoma are rare presenting 1 to 4, 7%of ovarian organictumors. These tumors are of stromal origin and contain varied proportion of fusiform connective tissue cells and theca cells. They mainly affect menopausal or perimenopausal women. This tumor is benign in most cases and may be responsible for hormone secretion. OBJECTIVES: Study the epidemiological and clinical data of patients with ovarian fibro-thecoma, analyze ultrasonographic characteristics of these tumors,and evaluate the sensitivity and specificity of pelvic ultrasound in ovarian fibro-thecoma approach. METHODS: A retrospective study of 47 patients who underwent surgical treatment for ovarian fibro-thecoma was performed. Data were collected in our department of gynecologyand obstetrics A within Charles Nicole hospital in Tunis, over a period of 18 years between January 1994 and December 2012. For each of our observations, we analyzed the clinical and para-clinical data, including U.S. characteristics and available MRI data with confrontation to the final histological results. RESULTS: The average age of patients was 45.2 years. The average gravidity was 4 and the mean parity was 3. . Thirty-eight of our patients were postmenopausal (80.85%). Ovarian tumor was discovered incidentally in 11 cases and on the occasion of functional symptoms in 36 cases including pelvic pain in 18 cases. Physical examination revealed a pelvic mass in 17 patients and pelvic-abdominal in 14 patients. All patients underwent a pelvic ultrasound. . Ultrasound identified 49 tumors (2 cases of bilateral tumors). Average size of tumors was 10, 05 cm (4 to 30 cm). ) . Ovarian tumor was echogenic in 9 cases (18.36%), hypoechoic in 14 cases (28.47%), mixed in 14 cases (28.47%) and anechoic in 12 cases (24.49%). The tumor was found to be solid in 27 cases (55.1%); cystic in 8 cases (16.3%) and solido cystic in 14 cases (28.6%).It was compartmentalized in 10 cases. Extra cystic vegetations were found in 2 patients. The tumor was nonvascularized at color Doppler in 47 cases (95.9%) and slightly vascularized in 2 cases (4.1%). Intra peritoneal effusion was objectified in 15 cases. The diagnosis of ovarian fibro-thecoma was raised based on U.S in 25 cases (51.02%) before surgery .MRI was performed in four cases. All patients underwent surgery. We performed laparotomy in 36 cases and laparoscopy in 11 cases. By laparotomy were performed a total hysterectomy with bilateral oophorectomy in most cases (26 patients). By laparoscopy we did lumpectomy in all cases. . The final pathologic examination revealed 19 fibromas, 14cystadénofibromas and 14 fibrothecomas. CONCLUSION: The paraclinical exploration of ovarian fibro-thecoma isbased, as all ovarian tumors, on ultrasound examination. The most typical features are images of solid tumors with regular contours, echogenic or mixed with the presence of streakedshadows.

High cellularity and mitotic activity in a primary ovarian fibro-thecomatous tumor of a young patient: a diagnostic and clinical challenge.

Purpose ofInvestigation: Solid ovarian tumors represent a clinical challenge, in particular in case of young patients who require a fertility sparing treatment. The authors report a case of hypercellular mitotically active ovarian fibrothecoma in a very young woman, successfully treated with a fertility sparing surgery. MATERIALS AND METHODS: A 21-year-old nulliparous woman presented at the present hospital with a 14-cm right ovarian mass, consisting of solid and pseudo-cystic components. There was neither an elevation of tumor markers nor evidence of metastatic disease. A laparotomic right salpingo-oophorectomy was performed. Uterus and left adnexa were preserved. RESULTS: The neoplasm consisted of a prevalent population of spindle-shaped elements and of a minor component of cells with wider cytoplasms, attributable to a thecomatous differentiation. The mitotic activity was focally elevated. Cytological atypia was mild to focally moderate. Clear areas of coagulative necrosis were not observed. At present 48 months after surgery, the patient is alive with no evidence of recurrence. CONCLUSIONS: The authors reported the lesion as a hypercellular and mitotically active fibrothecoma. The uneventful follow-up confirms the low malignant potential of the lesion. Caution is required reporting hypercellular stromal ovarian tu- mors, in order to avoid overdiagnosis and overtreatment, particularly in young patients.

Ovarian thecoma-fibroma groups: clinical and sonographic features with pathological comparison.

BACKGROUND: Ovarian thecoma-fibroma groups (OTFG) are uncommon sex cord-stromal neoplasms. The objective of the study was to demonstrate clinical and sonographic features of OTFG and compare with surgical histopathology. METHODS: A total of 61 patients with surgically proven OTFG were enrolled in this retrospective study to demonstrate its clinical and sonographic features and to compare with pathological findings. Gray scale and color Doppler sonography were performed presurgically with either transabdominal or transvaginal approach to image pelvic structures and lesions. The clinical findings and sonographic appearances were compared with the types of the OTFG tumors based on the histopathological diagnosis. RESULTS: The mean patient age was 53.57 (range, 26-86) years. There were 63.93% (39/61) patients in postmenopausal and 63.93% (39/61) patients with no clinical symptoms. Ultrasound findings of OTFG revealed as solid tumors with a typical feature of well-demarcated hypoechoic masses in 70.49% (43/61), among which 74.41% (32/43) tumors were smaller than 5 cm in diameter. There were 17 mixed echogenic masses with calcification, hemorrhage, or cyst, among which 70.59% (12/17) lesions were larger than 5 cm in diameter. Acoustic attenuation of the tumor was presented in 44.26% (27/61) of the cases. Doppler flow signals within the tumors were found in 20 cases (32.79%), in which 80% (16/20) had minimal or moderate flow signals. Ascites was detected in 32.79% (20/61) of the cases, Megi's syndrome was found in 1 case. Final pathology revealed 41 (67.21%) thecoma-fibromas, 15 (24.59%) fibromas, 4 (6.56%) thecomas and 1 (1.64%) fibrosarcoma. There were 58 patients underwent cancer antigen 125 (CA125) test, and 20.69% (12/58) showed an elevated level. The diameter of tumors was found to be significantly correlated with CA125 level (p < 0.01) and the amount of ascites fluid (p < 0.05). CONCLUSIONS: The typical sonographic features of OTFG include adnexal hypoechoic masses with clear border and acoustic attenuation as well as minimal Doppler flow signals. All the aforementioned features could make ultrasound imaging as a assistent tool improve the preoperative diagnostic accuracy.

Inhibin A and inhibin B producing ovarian fibrothecoma revealed by suppression of follicle stimulating hormone (FSH) in a post-menopausal woman: report of the first case.

In this report, we describe the first case ever reported in the literature, of an inhibin-A (INHA) and inhibin-B (INHB) producing fibrothecoma. A post-menopausal woman was referred to our unit because of follicle stimulating hormone (FSH) level below the reference interval for postmenopausal women. By contrast luteinizing hormone, hCG, and estradiol levels were within normal range. This discrepancy suggested the secretion of FSH inhibitory factors. INHB and INHA levels were markedly elevated for age, 475 pg/mL and 100 pg/mL, respectively. Ultrasonography and MRI showed a pelvic mass of indeterminate nature. Abnormal inhibin secretion is generally observed in granulosa cell tumors. In this case this etiology was unlikely because of low estradiol and AMH levels. Surgical exploration revealed a 10 cm mass of the left ovary proven histologically to be an ovarian fibrothecoma (OFT). After tumor removal, INHB and INHA levels decreased rapidly. Only three cases of OFT with an important secretion of INHB have been reported to date. INHA secretion has never been associated with OFT. There is a need to develop coupled hormone and imaging strategies to diagnose the source of INH secretion in case of FSH/LH discrepancy.

Fibrotecoma extraovárico gigante retroperitoneal / Giant Retroperitoneal Extraovarian Fibrothecoma

El fibrotecoma ovárico es una neoplasia poco frecuente y su localización extraovárica es más rara aún. Se observa generalmente como tumor sólido unilateral, de tamaño variable, en mujeres premenopáusicas. En su mayoría, es benigno y puede ser funcionales o no. Actualmente solo se han reportado tres pacientes y solo una de ellas de localización retroperitoneal. Se presenta un caso de una paciente con fibrotecoma extraovárico gigante retroperitoneal. Se realizó a la paciente exéresis de la lesión sin complicaciones intra ni posoperatorias.

Ovarian fibrothecoma is a rare malignancy and its extraovarian location is even rarer. It is generally seen as unilateral solid tumor, of variable size, in premenopausal women. It is mostly benign and it can be functional or not. Currently only three patients have been reported and only one of retroperitoneal location. A case of a patient with giant retroperitoneal extraovarian fibrothecoma is presented in this paper. The patient underwent excision of the lesion showing no intraoperative or postoperative complications.

Vestibulodynia: A Multifactorial Syndrome: A Case Report.

BACKGROUND: Vulvodynia is a difficult-to-treat, chronic, multifactorial malady that drastically lowers the quality of life of afflicted patients. CASE: A 68-year-old woman, who had been treated successfully for vulvodynia years before with medication, returned with a recurrence of vulvodynia symptoms that this time did not respond to treatment. She now had biopsy-confirmed lichen sclerosis and was found to have markedly elevated serum testosterone levels. An imaging study detected an ovarian lesion that, on removal, proved to be afibrothecoma. Postoperatively the testosterone rapidly dropped to normal levels. What was unexpected and unusual was that the vulvar pain disappeared and the lichen sclerosis markedly regressed. CONCLUSION: This case demonstrates a hormonal trigger for the development of vulvodynia.

Accuracy of intra-operative frozen section and its role in the diagnostic evaluation of ovarian tumors.

UNLABELLED: Summary OBJECTIVE: This retrospective study was undertaken to evaluate the accuracy and role of intra-operative frozen section in the diagnosis of ovarian tumors. MATERIALS AND METHODS: Retrospective study of 804 ovarian frozen section results between June 2010 and June 2014 was examined to determine the accuracy of frozen section diagnosis. The intra-operative frozen section diagnosis was compared with the permanent (paraffin) section and the overall accuracy, sensitivity, specificity, and positive and negative predictive values of the frozen section were studied. RESULTS: The overall accuracy to determine the status of malignancy was 92.6%. There were 38 (7.4%) false negative and no false positive frozen section diagnoses.The sensitivity, specificity, and positive predictive and negative predictive values for benign ovarian tumors were 100.0%, 97.0%, 91.3%, and 100.0%, respectively; for borderline tumors they were 64.3%, 97.0%, 91.5%, and 94.0%, respectively, and for malignant tumors they were 90.0%, 100.0%, 100.0%, and 85.5%, respectively. CONCLUSION: This study concluded that frozen section appears to be an adequate technique for the histopathological diagnosis of ovarian tumors, with some limitations observed among borderline and mucinous tumors.

Evidence of Spontaneous Recovery of Granulosa-Theca Cell Tumour in a Heifer: A Retrospective Report.

A 14-month-old Japanese Black heifer was evaluated on Day 0 (D 0) for enlargement of the right ovary (RO). Transrectal ultrasonography (TRUS) revealed that the RO was markedly enlarged and multicystic, while the left ovary (LO) was small and inactive. The presumptive diagnosis was granulosa-theca cell tumour (GTCT), which was confirmed by markedly elevated plasma anti-Müllerian hormone (AMH) of 4.42 ng/ml. Therefore, ovariectomy of the RO was the treatment of choice. The heifer was checked by TRUS and blood sampling on D 42, D 63 and immediately before ovariectomy on D 85. On D 42, TRUS did not show marked changes in either ovary in comparison with D 0. However, on D 63, the RO had transformed into a single cyst, and on D 85, the LO had resumed cyclic activity. The RO was extracted on D 85 by hand-assisted laparoscopic ovariectomy to allow better control. Unexpectedly, histopathology revealed the lesion to be a fluid-filled cystic structure, with no neoplastic proliferation of follicular epithelium that would indicate GTCT. The wall of the cystic structure consisted of collagen fibres and a few degenerated granulosa cells. The retrospective hormonal analysis revealed that the AMH concentrations had markedly dropped on D 63 and 85, which coincided with resumption of cyclicity in the LO. These findings suggest that the GTCT had self-cured and transformed into a cyst-like structure. The heifer then received an oestrous synchronization regime on D 105, was artificially inseminated on D 115 and became pregnant.

[Ovarian tumor in a koi carp (Cyprinus carpio): Diagnosis, surgery, postoperative care and tumour classification]. / Ovarialtumor bei einem Koi (Cyprinus carpio): Diagnose, chirurgische Therapie, Nachsorge und Tumordiagnostik.

Although ovarian tumour in the koi (Cyprinus carpio) does not appear to be an uncommon condition, its occurrence and therapy has rarely been reported. In the present case, the decision for surgery was based on clinical and sonographic findings of an intracoelomic mass. We used tricaine methansulfonate for the anaesthesia. Laparotomy was performed by ventral access and an ovarian tumour of 12-cm diameter was removed. The wound was sutured in two layers using Vicryl®. In addition to the application of an analgesic, an antibiotic and vitamins, the postoperative conditions the patient was kept under were adapted to support wound healing. The fish recovered uneventfully and was clinically healthy during the 16-month observation period. Based on the histological findings, the tumour was diagnosed as a thecoma. Investigations using antibodies against vimentin, cytokeratin, S 100 and glial fibrillary acidic protein (GFAP) failed to provide reliable results.