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A 65-year old woman presents with a Demons-Meigs syndrome characterized by dyspnea resulting from a transsudative pleural effusion, an important unilateral right ovarian mass and ascites. The diagnosis of a Brenner type histology was obtained after complete surgical removal of ovarian tumor. After discharge the patient entered in a sustained complete response and thus potential cure. Brenner tumor is a rare and often benign ovarian affection. The clinical signs aren't generally much specific: pelvic pain or heaviness, metrorrhagia and menstrual irregularity may be observed. Brenner tumor may exceptionally induce a Demons-Meigs's syndrome. This syndrome associates one or more benign tumors of the female reproductive tract with pleural and peritoneal effusions. This could depict a rich but disturbing clinical picture. The prognosis and the regression of the symptomatology are nevertheless excellent after tumor surgical resection.

Borderline Brenner tumors represent quite a rare entity of ovarian tumors (about 2%) that develop from the surface ovarian epithelium. They are formed from papillary structures made of fibrovascular conjunctive axes covered by a transition epithelium, similar to the urinary bladder epithelium. According to the WHO classification, Brenner tumors present the following forms: benign, borderline and malignant. The benign ones are the most frequent, representing about 95%, the borderline represent about 5%, and the malignant ones less than 1%. We present the case of a 64-year patient who was diagnosed with right ovary cyst. The histopathological examination highlighted the presence of a borderline Brenner tumor at the same time with the cystic lesion, on the same ovary. The surgical treatment led to a complete cure of the patient, so that the yearly ultrasound reexamination did not trace the presence of any tumoral relapse.

Ovarian Sertoli-Leydig cell tumours (SLCT), also termed arrhenoblastomas, are the most frequent virilising tumours in women of reproductive age. Very rare secretory Brenner tumours (BT) have been described, generally after the menopause. A 31-year-old woman sought medical advice for secondary amenorrhoea, progressive hirsutism and a 5-year history of virilisation syndrome with clitoromegaly. Testosterone was markedly high (285 ng/dl, N<85) with moderate elevation of delta 4-androstenedione (D4AD) (311 ng/dl, N <270), dehydroepiandrosterone sulfate (DHEAS) (366 μg/dl, N <340) and 17-hydroxyprogesterone (17OHP) (275 ng/dl). LH was 9 IU/l, FSH 4.3 IU/l, estradiol 60 pg/ml and progesterone 314 ng/100 ml. Cortisol was decreased (1.3 μg/dl) after the dexamethasone suppression test. Pelvic MRI showed a 5-cm right ovarian tumour with a 2.5 cm nodular component and cystic areas, and two nodules measuring 11 mm and 15 mm above the right and left ovaries. After right ovariectomy by laparoscopy, pathological examination concluded on a 3-cm SLCT and a 2-cm BT; the nodules above the ovaries were dysembryoplastic cysts. Postoperatively, testosterone level was normal after 24 h (26 ng/dl), estradiol and progesterone rapidly decreased, cyclic secretion then resumed and the patient menstruated at day 27. To our knowledge, this is the first report of an ovarian tumour associating a Sertoli-Leydig cell tumour and a Brenner tumour in a patient with virilisation syndrome which resolved after ovariectomy.

This case report describes the onset of hypertension and virilisation during pregnancy due to a Brenner tumour., (© 2010 The Authors. Australian and New Zealand Journal of Obstetrics and Gynaecology © 2010 The Royal Australian and New Zealand College of Obstetricians and Gynaecologists.)

Transitional cell carcinoma of the endometrium (TCCE) is a subtype of endometrial carcinoma, characterized by a prominent papillary pattern, resembling the papillary carcinoma of the urothelium. This neoplasm is very rare, with only 13 cases reported in the international literature. In this paper, a new case of TCCE associated with benign ovarian Brenner tumor is described. This association is extremely rare, with only 1 other case reported. A review of the literature is performed to delineate the clinico pathologic features of this malignancy. Moreover, immunohistochemical and molecular studies are carried out in the effort to establish the phenotype and etiology of this rare neoplasm. The molecular study, by polymerase chain reaction (PCR) failing to reveal the presence of HPV DNA, demonstrates that neither the TCCE nor the ovarian Brenner tumor is caused by an HPV infection. The association of TCCE with benign ovarian Brenner tumor could be a coincidental event. Conversely, this finding could be the manifestation of a multicentric metaplastic process (neometaplasia), involving both the coelomic epithelium of the ovary and the Mullerian epithelium of the uterus, or the evidence of "field effect" that manifests differently at different anatomical sites. In our view, other cases of TCCE associated with ovarian Brenner tumor should be reported to confirm the last 2 hypotheses.

Although there are several reports of Brenner tumor showing estrogen activities, it is an extremely rare cause of androgen excess leading to virilism, and the source or mechanism of its androgen production is also unknown at present. A 74-year-old woman presented with lower abdominal pain and increased facial hair growth of 6-month duration. Bilateral ovarian tumors were detected, and her serum testosterone (1.7 ng/mL) and estradiol (75 pg/mL) levels were elevated. Bilateral salpingo-oophorectomy was performed. The ovarian tumors were diagnosed as benign Brenner tumor associated with fibrothecoma-like and luteinized stromal cells. Postoperatively, the serum testosterone and estradiol levels decreased. Immunohistochemically, fibrothecoma-like stromal cells were positive for cytochrome P-450 aromatase, which catalyzes the conversion from androgen to estrogen, and negative for c-Jun protein, which has recently reported to attenuate estrogen biosynthesis by directly down-regulating transcription of the aromatase gene. On the other hand, luteinized stromal cells were negative for cytochrome P-450 aromatase and positive for c-Jun protein. It is suggested that androgen is produced mainly in the luteinized stromal cells, because androgen is not converted to estrogen caused by suppression of aromatase biosynthesis by c-Jun.

Background: There has been controversy regarding the histogenesis of Brenner tumors. It is generally accepted that Brenner tumors are derived directly from ovarian surface epithelium, which undergoes metaplasia to form the typical urothelial-like components, whereas some investigators assume that Brenner tumors arise from immature germ cells., Case: We describe a well-documented case of the coexistence of struma ovarii regarded as a form of teratoma and Brenner tumor in the same ovary. Immunohistologically, not only columnar cells of thyroid follicles, but also transitional cells of Brenner nests were positive for thyroglobulin., Conclusions: In the present case, Brenner tumors and thyroid elements coexisted and were positive for thyroglobulin. While there is strong evidence that pure Brenner tumors originate mostly from the ovarian surface, at least Brenner tumors associated with teratomatous elements may have a germ cell origin.

Background: Meigs' syndrome refers to solid, benign ovarian tumors, ascites, hydrothorax, and resolution of these signs after surgery. Meigs' syndrome with an elevated CA 125 secondary to benign Brenner tumors is exceedingly rare., Case: A postmenopausal woman presented with a large pelvic mass, ascites, and a right pleural effusion. Serum CA 125 was 759 IU/mL. Ascitic fluid, pleural fluid, and fine needle aspiration of the mass were without evidence of malignancy. Exploratory laparotomy with total abdominal hysterectomy and bilateral salpingo-oophorectomy revealed benign Brenner tumors. Immunohistochemical staining for CA 125 showed immunoreactivity in the omentum only. Postoperatively, her signs and symptoms resolved completely and did not recur., Conclusion: Cytologic or histologic confirmation of malignancy is imperative in patients with a pelvic mass, ascites, hydrothorax, and elevated CA 125 before initiating chemotherapy.

Background: The role of thrombospondin (TSP) in tumor progression remains controversial. The association of TSP with clinicopathological features regarding prognostic significance was examined in patients with epithelial ovarian tumor., Materials and Methods: Gene expression of TSP-1 and TSP-2 was assessed by reverse transcriptase-polymerase chain reaction in 6 borderline and 29 malignant epithelial ovarian tumors., Results: TSP-1 mRNA expression was detected in 14 out of the 29 malignant epithelial ovarian tumors (48.3%), whereas TSP-2 mRNA expression was detected in 7 malignant epithelial ovarian tumors (24.1%). In contrast, no specimen from the borderline epithelial ovarian tumors expressed TSP mRNA. TSP-1 expression was significantly higher in tumors with advanced stage, massive ascites, positive peritoneal cytology and high grade. TSP-2 expression was significantly higher in tumors with massive ascites. Patients exhibiting TSP-1 and -2 mRNA expression demonstrated a markedly poorer prognosis than those lacking TSP-1 and -2 mRNA expression., Conclusion: These findings provide evidence that TSP expression may be associated with an aggressive phenotype in this class of neoplasm.

Magnetic resonance imaging features were assessed in a benign Brenner tumor with an ipsilateral simple cyst. Brenner tumor showed isointensity to the uterine muscle on T1-weighted images and markedly low signal intensity on T2-weighted images. On the other hand, on T1-weighted images after administration of gadopentetate dimeglumine a Brenner tumor showed mild patchy enhancement. The low signal intensity seen on T1- and T2-weighted images may have been due to the abundant dense fibrous tissues.

A case of malignant Brenner tumor (MBT) in a 42-year-old woman is described. According to the literature it is evident how rare this type of MBT is as well as the histogenetic problems arising in this neoplasia deriving from the coelomic epithelium. The malignant histologic criteria are those adopted in 1973 by Hull and Combell who showed as unquestionable signs the association of benign and malignant Brenner tumor, the invasion of the stroma on behalf of the malignant Brenner elements and the absence of mucinous cystadenoma or the association with the above mentioned.

A 64-year-old woman with a 45,X karyotype and Turner's syndrome and recent onset of right lower quadrant pain was found to have a mucinous cystadenoma with focal areas of borderline malignancy and a typical Brenner tumor in the right adnexa. Ovarian tumors in patients with a 45,X karyotype and Turner's syndrome are extremely rare. The present paper is the first report of concurrence of these 2 tumors in an affected individual. The simultaneous occurrence and intimate admixture of these tumors in a patient believed to be devoid of germ cells would seem to favor a common histogenesis from the germinal epithelium.

Although the first Brenner tumor was reported in 1898 by MacNaughton-Jones, it was not until 1907 that Fritz Brenner described the tumor that bears his name. Since then more than 500 cases have been reported with bilaterality ranging from 3.7 to 8.0 per cent. However, this percentage is probably high. The tumor was originally thought to arise from follicular epithelium. Several other etiologies of the Brenner tumor have been suggested, including celomic surface epithelium, Walthard rests, teratoma, urothelium, and rete ovarii. The majority of Brenner tumors are inert, but there have been reports suggesting endocrine activity. The question of malignant transformation of the Brenner tumor has gained attention since Von Numers described the first malignant Brenner tumor in 1945. The determination of malignancy of these tumors has been hampered by vague histologic criteria for malignancy. The confusion surrounding the histogenesis, hormonal capabilities, and malignant potential of the Brenner tumor has persisted. A case report of bilateral Brenner tumors of the ovaries is presented as well as a review of the literature with emphasis on the bilaterality of Brenner tumors.

It will be reported about 33 cases of Brenner-tumors, observed between 1932 to 1974. Symptomatic, therapy, progress, morphology of these tumors as well as their coincidence with other pathological changes on genitals will be demonstrated. A short casuistic description of 3 cases with proliferating and 1 case of malignant Brenner-tumor will be given. In the discussion, it will be informed about the frequency, age, diagnostic and prognostic problems of proliferating and malignant tumors. The problem of hormonal activity of some of Brenner-tumors and finally the modern views over histogenesis will be considered and discussed.

A case of ovarian Brenner tumor displaying androgenic activity and clinical masculinization is reported in a 65-year-old woman. Plasma androstenedione, dehydroepiandrosterone sulfate (DHEAS), testosterone, dihydrotestosterone, cortisol, estradiol, FSH, LH, and prolactin were measured before and after suppression with dexamethasone and stimulation with hCG, and again after removal of the tumor. The plasma androgenic compounds were measured in both ovarian and peripheral veins. Basal levels of androstenedione and of total testosterone were significantly elevated, decreasing to normal ranges after operation and remaining after 3 months. There was a sharp increase of total testosterone, DHEAS, and dihydrotestosterone levels after administration of hCG. The levels of androstenedione, DHEAS, testosterone, and dihydrotestosterone were higher in the ovarian vein than in the peripheral vein. These findings indicate that the tumor was the main source of these androgenic hormones.