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.

Meigs' syndrome with elevated serum CA 125 level in a case of ovarian fibrothecoma.

Meigs' syndrome is the association of benign ovarian tumor, pleural effusion, and ascites. Meigs' syndrome with marked elevated CA 125 is a rare clinical entity and only 42 cases have been reported. Although there is difficulty in discerning the diagnosis of Meigs' syndrome from that of an ovarian malignancy, it should be considered in the differential diagnosis in postmenopausal patients with an ovarian mass, hydrothorax, ascites, and elevated CA 125. In this report, the authors present the case of a 52-year-old postmenopausal woman with ovarian fibrothecoma, pleural effusion, ascites, and elevated CA 125 (319.2 IU/ml). Exploratory laparotomy with total hysterectomy and bilateral salpingo-oophorectomy was performed, and the pathologic diagnosis was ovarian fibrothecoma. After the surgery, the pleural effusion disappeared spontaneously and the CA 125 became normal. The authors also summarized other cases of Meigs' syndrome with elevated CA 125, and reviewed the mechanism of elevation of CA 125, ascites, and pleural effusion.

[Comparision of sonographic findings and pathological features in ovarian thecoma].

OBJECTIVE: To compare the sonographic findings with pathological features of ovarian thecoma, and to analyze the relationship between them. METHODS: The sonograms of 45 ovarian thecoma cases were reviewed retrospectively and categorized into three subtypes as sound attenuation pattern, homogeneous hypoechoic pattern and solid and cystic mixed pattern. The pathological findings were classified as theca cell-predominant, fibroblast-predominant and mixed thecoma according to the cellular composition of the tumors. Hyaline degeneration and luteinization of the tumors were recorded. The pathologic findings of each subtype based on sonography were compared. RESULTS: Of the 45 patients, there were 34 (75.6%) solid ovarian lesions, 15 (33.3%) of those showed a sound-attenuation pattern with an anterior hypoechoic zone and posterior acoustic attenuation in sonography, the other 19 (42.2%) cases had homogeneous hypoechoic pattern with no posterior acoustic attenuation, and the remaining 11 (24.4%) cases presented as a solid and cystic mixed pattern. There were no significant differences in pathological cellular composition among the three sonographic subtypes. Five solid tumors containing hyaline degeneration and one with luteinization were found to have posterior acoustic attenuation. The solid and cystic mixed thecomas showed cystic degeneration and hemorrhage. CONCLUSION: Solid ovarian thecomas usually have typical sonographic features, which may be associated with degeneration but not with cellular composition within the tumor.

Bilateral occurrence of granulosa-theca cell tumors in an Arabian mare.

An Arabian mare was referred for right granulosa-theca cell tumor (GTCT) evaluation. The mare was presented 4.5 years later for a left GTCT, after successfully conceiving and delivering a normal foal in the interim. The concurrent or nonconcurrent occurrence of bilateral GTCT in mares appears to be rare.

Discriminating between virilizing ovary tumors and ovary hyperthecosis in postmenopausal women: clinical data, hormonal profiles and image studies.

BACKGROUND: The presence of virilizing signs associated with high serum androgen levels in postmenopausal women is rare. Virilizing ovarian tumors (VOTs) and ovarian stromal hyperthecosis (OH) are the most common etiologies in virilized postmenopausal women. The differential diagnosis between these two conditions is often difficult. OBJECTIVE: To evaluate the contribution of clinical features, hormonal profiles and radiological studies to the differential diagnosis of VOT and OH. DESIGN: A retrospective study. SETTING: A tertiary center. MAIN OUTCOME MEASURES: Clinical data, hormonal status (T, E2, LH and FSH), pelvic images (transvaginal sonography and MRI) and anatomopathology were reviewed. PATIENTS: Thirty-four postmenopausal women with a diagnosis of VOT (13 women) and OH (21 women) were evaluated retrospectively. RESULTS: Clinical signs of hyperandrogenism were more prevalent in the VOT group than the OH group. Although the VOT group showed higher T and E2 levels and lower gonadotropin levels than the OH group, a great overlap occurred among the hormone levels. A pelvic MRI provided an accurate differentiation of these two conditions. CONCLUSION: In this group of patients, the main features contributing to the differential diagnosis of VOT and OH were serum levels of testosterone and gonadotropins and the presence of an ovarian nodule identified on the MRI. Although the association of clinical, hormonal and radiological features contributes to the differential diagnosis of these two conditions, histopathological analysis remains the gold standard for the diagnosis of ovarian hyperandrogenism in postmenopausal women.

Serum 17 alpha-hydroxyprogesterone in patients with gestational trophoblastic neoplasms.

Serum 17alpha-hydroxprogesterone (17-OHP), progesterone (P), and human chorionic gonadotropin (hCG) levels were measured by specific radioimmunoassay in 19 patients undergoing laparoscopy or laparotomy with either unevacuated molar pregnancy or nonmetastatic gestational trophoblastic neoplasms (GTN), in 10 normal pregnant patients at equivalent gestational age (7-21 weeks), and in 4 patients with metastaic GTN following hysterectomy and bilateral salpingo-oophorectomy. All patients with theca lutein cysts had significantly elevated serum 17-OHP levels compared to those in 1) normal pregnancy, 2) patients with GTN and normal-size ovaries, 3) patients with metastatic GTN in the absence of ovaries (P less than 0.02). Levels of serum 17-OHP but not P correlated with the degree of ovarian enlargement (r = 0.87, P less than 0.05). Serum P concentrations in patients with theca lutein cysts, although higher than the levels in cases of GTN with normal-size ovaries, were not significantly different from the levels in normal pregnancy (P greater than 0.05). Serum hCG levels in patients with theca lutein cysts, though higher than the normal pregnancy levels (P less than 0.05), were not significantly different from those in cases of GTN with normal-size ovaries and GTN without ovaries (P greater than 0.05). Under the conditions studied, no correlation was observed between serum hCG and P levels in our cases of GTN. Increased serum 17-OHP level in a patient with GTN suggests the presence of theca lutein cysts.

Accuracy of tissue polypeptide specific antigen (TPS) in the diagnosis of ovarian malignancy.

This paper evaluates the usefulness of determining the level of serum Tissue Polypeptide Specific Antigen (TPS) by TPS ELISA in the diagnosis of ovarian malignancies and compares it with the results of histological examination of the ovaries. The study covers 92 patients who had been clinically diagnosed with ovarian tumors by history, physical examination and pelvic examination with or without pelvic ultrasonography. All of them underwent surgical treatment by exploratory laparotomy in Siriraj Hospital between May 1, 1996 and 31 March, 1997. None of the patients had been treated with chemotherapy or hormonal therapy, and in no case were there any previously diagnosed malignancies. TPS was measured in the serum of 92 patients who were preoperatively diagnosed with ovarian tumor: 52 patients had benign pelvic masses and 40 patients had malignant ovarian tumors. Using the criterion for TPS positivity defined by the manufacturer (80 U/L), TPS levels were elevated in 28.8% of benign pelvic mass patients and in 90% of malignant ovarian tumor patients. Statistical analysis using a two-by-two table at every cut-off TPS level and a receiver operating characteristic (ROC) curve, the optimal accuracy, sensitivity, specificity, positive predictive value, negative predictive value, false positive rate and false negative rate were 79.4, 90.0, 71.2, 70.6, 82.9, 28.9 and 10.0%, respectively using the positivity criterion of 80 U/L. We conclude that determination of serum TPS level by TPS ELISA in the diagnosis of ovarian malignancy is good and clinically acceptable. A TPS level greater than 80 U/L is a useful positivity criterion for screening for malignant ovarian tumors, while a TPS level greater than 180 U/L is a positivity criterion for differentiating malignant ovarian tumor from benign pelvic mass. Because of its high false positive rate, any patient with TPS greater than 80 U/L should be further investigated for malignant ovarian tumor.

Endocrinological changes before and after removal of the granulosa theca cell tumor (GTCT) affected ovary in 6 mares.

To clarify the endocrinological characteristics of the mares with granulosa theca cell tumor (GTCT), peripheral plasma samples from the 6 mares affected with GTCT were collected before and after the surgical removal of the affected ovary. Concentrations of testosterone (T), follicle stimulating hormone (FSH), luteinizing hormone (LH), immunoreactive-inhibin (ir-INH), progesterone (P) and estradiol-17beta (E(2)) in the plasma samples were measured by radioimmunoassay. Before removal of GTCT in all cases, the concentrations of T were significantly higher than those of normal mares at the breeding and non-breeding seasons, whereas plasma concentrations of FSH, LH, ir-INH, P and E(2) were lower. After surgical removal of the affected ovary, the circulatory concentrations of T was declined, but the concentrations of other hormones were constantly low as compared with those of normal mares. The present study suggests that 1) the source of higher T may be due to the abnormal follicles in ovary of GTCT, 2) in the case of GTCT the elevated level of T is observed due to the lack of aromatase, and 3) the high level of T is a typical characteristics for GTCT in mares. It is also suggested 4) due to the elevated levels of T the concentrations of gonadotropins may be suppressed.

Meigs' syndrome with elevated CA 125: case report.

CONTEXT: Meigs' syndrome consists of a benign ovarian tumor accompanied by ascites and hydrothorax. Elevated serum CA 125 levels in postmenopausal women with solid adnexal masses, ascites and pleural effusion are highly suggestive for malignant ovarian tumor. However, patients with Meigs' syndrome can also have elevated serum CA 125 levels. The authors report a case of Meigs' syndrome with elevated CA 125 level. OBJECTIVE: This is a case report of Meigs' syndrome with elevated CA 125 level. CASE REPORT: A 65-year-old Brazilian woman had presented progressive dyspnea, weight loss and decline in general condition over the 7 months preceding admission to our service. In another hospital, the patient had been submitted to thoracic drainage due to pleural effusion. With recurrence of the pleural effusion and increase in abdominal volume due to ascites and a pelvic mass, the patient sought our service. Transvaginal ultrasound showed an extensive adnexal solid mass of 16.4 x 10.8 cm located in the pelvis without exact limits, and the serum CA 125 level was elevated. With a preoperative diagnosis of ovarian carcinoma, the patient was submitted to exploratory laparotomy, which revealed a left ovarian tumor. The frozen section diagnosis was thecoma. Total abdominal hysterectomy with bilateral salpingo-oophorectomy was performed. The histology of the specimen confirmed the diagnosis of thecoma. The patient was asymptomatic with a normal serum CA 125 level 20 months after the operation.

[Virilism during pregnancy]. / Virilisme au cours de la grossesse: à propos d'un cas.

We discuss the causes of virilism in pregnancy and the impact of hyperandrogeny on the female foetus. We report a case of virilism in a 28-year-old, gravida 1, para 1 patient with normal pregnancy and review the literature. After conception, the patient had been well until the 18th week of gestation, when she developed signs of virilization; her serum testosterone was markedly elevated. She delivered of a normal male infant at term. After delivery, signs of virilization regressed with normalization of testosterone level but a wide mass of the left ovary that persisted for 8 months. The histological study of the tumor showed luteinized thecoma. Luteomas and hyper-reactio luteinalis were the principal causes of virilism in pregnancy, thecomas are rare.

[Serum estradiol as a marker in recurrent granulosa-theca-cell tumor]. / Serumøstradiol som markør ved recidiverende granulosa-thecacelltumor.

A case of a granulosa-theca-cell tumour of the ovary in a 39-year-old woman is reported. Despite benign histology and apparently radical operation, tumor growth relapsed twice before apparent cure. Oestradiol in serum proves valuable as a tumour marker and correlates well with tumour relapse.

[Case report of a granulosa-theca tumor in a cow]. / Case study van een granulosa-thecaceltumor bij het rund.

A 2 year-old cow with abnormal behaviour was observed during a farm visit. Rectal palpation of the cow revealed the presence of a mass of at least 12 cm in diameter. After further examination, it appeared that 'ovarian tumour' was the most likely differential diagnosis. In order to confirm this diagnosis, blood samples were drawn and analysed for plasma progesterone and plasma oestradiol-17 beta concentrations. Also, the gross pathology and histology of the mass were evaluated. The combination of the clinical presentation of the cow, the hormone concentrations, and the histological appearance of the mass confirmed the diagnosis ovarian tumour. The tumour was classified as granulosa-theca cell tumour.

Ovarian thecoma with virilizing manifestations.

A 29-year-old woman presented with secondary amenorrhea, primary infertility, and virilization, which had developed over the past 2 years was suspected to have a virilizing tumor at her left ovary. Her serum testosterone level was markedly elevated (380 ng/dL). Left salpingoophorectomy was performed, and histopathological examination revealed a thecoma of the left ovary. The postoperative serum testosterone level returned to 65 ng/dL. The patient did not have regression of virilism soon. However, the patient had a normal menstruation 29 days after surgery and gave birth to a baby 13 months after surgery.

The significance of serum progesterone and serum unconjugated oestradiol-17beta in unaborted hydatidiform mole.

Both serum progesterone and serum unconjugated oestradiol-17beta (Oe2) were measured by competitive protein binding assay and radioimmunoassay respectively in 42 cases of unaborted hydatidiform mole. Serum human chorionic gonadotrophin (HCG) was measured by a haemagglutination-inhibition technique. In 26 cases of intact molar pregnancies without theca lutein cysts (TLC), serum progesterone ranged from 18.0 to 289.0 ng/ml with a mean +/- standard error of the mean (SEM) of 65.9 +/- 13.1 ng/ml; serum Oe2 ranged from 4.0 to 37.0 ng/ml with a mean +/- SEM of 17.9 +/- 1.9 ng/ml; serum HCG ranged from 60 to 1920 IU/ml with a mean +/- SEM of 531.5 +/- 105.7 IU/ml. In contrast, 16 cases of intact molar pregnancies with TLC had serum progesterone ranging from 34.1 to 288.0 ng/ml with a mean +/- SEM of 134.1 +/- 2.4 ng/ml; serum Oe2 ranging from 1.7 to 76.3 ng/ml with a mean +/- SEM of 31.5 +/- 5.3 ng/ml; serum HCG ranging from 320 to 2560 IU/ml with a mean +/- SEM 1400 +/- 196.2 ng/ml. The differences between the mean of these three hormones in hydatidiform mole with and without TLC were significant (progesterone: P less than 0.005; Oe2: P less than 0,0125; HCG: P less than 0.005). There was a significant correlation between serum HCG and serum Oe2 (coefficient of correlation r = +0.3565, P less than 0.0125) and between serum Oe2 and serum progesterone (r = +0.3787, P less than 0.0125). There was no significant difference in the mean levels of serum progesterone, Oe2 and HCG in hydatidiform mole with and without subsequent malignant sequelae. The mean ratios of Oe2/progesterone were essentially similar in moles with and without TLC and with and without malignant sequelae. The significance of these findings are discussed.

Human chorionic gonadotropin, estriol, and testosterone changes in two pregnancies with hyperreactio luteinalis.

A case of hyperreactio luteinalis is presented with measurements of human chorionic gonadotropin, total and free testosterone, and estriol throughout two term pregnancies. Ovulation induction with menotropins resulted in one spontaneous first-trimester abortion, one singleton term pregnancy, and 1 triplet pregnancy that converted to a singleton term pregnancy after spontaneous degeneration of two fetuses in the first trimester. During the first term pregnancy, levels of human chorionic gonadotropin peaked at 34 weeks of gestation (169,000 mIU/ml) and levels of total testosterone peaked at the same time (1777 ng/dl). Free testosterone (16.6 ng/dl) and estriol (10.8 ng/ml) reached their peak at 36 weeks of gestation. During the second term pregnancy, peak values of human chorionic gonadotropin (150,900 mIU/ml) and total testosterone (870 ng/dl) occurred at 10 and 5 weeks of gestation, respectively, before two fetuses of a triplet pregnancy degenerated.