Cases reported "Virilism"

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1/37. Diffuse stromal Leydig cell hyperplasia: a unique cause of postmenopausal hyperandrogenism and virilization.

    A 60-year-old woman presented with diffuse scalp alopecia, hirsutism, and clitorimegaly, and the mean serum testosterone levels were greater than 200 ng/dL. Findings on computed tomography of both adrenal glands were normal. After bilateral oophorectomy, a unique histological picture consisting of diffuse stromal Leydig cell hyperplasia was found. Reinke crystals were present, but neither hilus cell hyperplasia nor stromal hyperthecosis was noted. Sequencing of the 11 exons of the gene for the luteinizing hormone receptor revealed no abnormality. Relevant data suggest that treatment of the postmenopausal woman with hyperandrogenism and virilization is bilateral laparoscopic oophorectomy if she has no pronounced ovarian enlargement or adrenal tumor on imaging. In this setting, an intensive endocrine evaluation or a search for metastatic disease seems to be unnecessary.
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ranking = 1
keywords = lutein
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2/37. Virilization in bilateral macronodular adrenal hyperplasia controlled by luteinizing hormone.

    We report a case of a virilized 59-yr-old woman with elevated serum testosterone levels and bilateral macronodular adrenal hyperplasia. The patient underwent laparoscopic right adrenalectomy, after which the elevated testosterone level transiently normalized. The immediate postoperative depression of the testosterone level suggested that the process was driven by gonadotropins that were suppressed by the stress of surgery. The excised right adrenal mass contained testosterone by immunohistochemistry and LH receptor mRNA by in situ hybridization. The recurrence of hyperandrogenemia suggested that the enlarged left adrenal was also secreting testosterone. The serum testosterone level increased in response to im injection of human chorionic gonadotropin, suggesting control by aberrant LH receptors. Injection of leuprolide acetate (7.5 mg im) to suppress LH levels resulted in normalization of the testosterone level 12 d later that persisted for several weeks. Ectopic receptors mediating Cushing's syndrome have been described in several cases of bilateral adrenal hyperplasia and adrenal adenoma. This is the first case to our knowledge in which pure androgen overproduction in adrenal hyperplasia has been shown to be controlled by LH receptors. In our patient, the control of androgen secretion by LH may explain the postmenopausal onset of virilization and the transient postoperative normalization of the serum testosterone level.
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ranking = 4
keywords = lutein
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3/37. Recurrent maternal virilization during pregnancy caused by benign androgen-producing ovarian lesions.

    Benign causes of maternal virilization in pregnancy, such as luteoma of pregnancy and hyperreactio luteinalis, are generally believed to resolve completely post partum and not to recur. We present the fifth case in the literature of recurrent maternal virilization in pregnancy. These lesions should be viewed as potentially recurrent and cases should be managed accordingly.
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ranking = 1
keywords = lutein
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4/37. Enzyme histochemistry of ovarina lipoid cell hyperplasia in a masculinized patient.

    In an effort to better identify the specific steroids produced by histologically similar cells of certain ovarian tumors, a battery of histochemical assays was performed on a masculinizing tumor removed from a 21-year-old patient. Certain reactions were distinguished that seemed to be specific for the histologically classic luteinized stromal cell, and these reactions can be correlated with those obtained with Leydig and hilar cells. The apparent precursors of the interstitial cells (ISC) of this tumor, and to a lesser extent the immature ISC, possess unusually high cholinesterase activity, especially with the butyryl ester. The importance of the perivascular and neural elements in the process of steroidogenesis is suggested by the findings in this case.
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ranking = 1
keywords = lutein
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5/37. Mucinous cystadenoma of the ovary with functioning stroma and virilization in pregnancy: a case report and review of the literature.

    Virilization caused by ovarian tumors with functioning stroma during pregnancy is extremely rare and has been reported in many ovarian tumors. In mucinous cystadenomas with maternal virilization during pregnancy the stromal cells responsible for the hormone secretion resemble lutein or leydig cells and have been referred to as luteinized stromal cells. We present a case of a 30-year-old, gravida 2, para 1, woman who presented at approximately the 38th week of pregnancy with features of virilization. At the same time, a cesarean section was performed because of fetal distress and a male weighing 3,030 g without any gross abnormalities was delivered. A large tumor of the right ovary was detected and a right salpingo-oophorectomy was performed. Histopathologically, the tumor proved to be a benign mucinous cystadenoma. Masses typically resembling lutein stromal cells or leydig cells of the testes or ovarian hilus were found in the wall of the cyst below the mucinous epithelium. No crystalloids of Reinke were identified. The stromal component of the tumor was characterized as functioning stroma with luteinized stromal cells. The glandular mucinous epithelium showed focal positivity for human chorionic gonadotrophin. The cytoplasm of the luteinized stromal cells reacted strongly and diffusely with antiserum for vimentin. Also, the cytoplasm of the luteinized stromal cells showed focal intense positivity for synaptophysin, and focal mild positivity for human chorionic gonadotrophin. Staining results for oestrogen and progesterone receptors were negative. In conclusion, we present an unusual case of clinical virilization during pregnancy associated with an ovarian mucinous cystadenoma with functioning stroma. The virilizing manifestations disappeared after removal of the ovarian neoplasm, supporting the perception that the functioning ovarian stroma was responsible for the androgen production.
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ranking = 6
keywords = lutein
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6/37. Virilisation in a case of transitional cell carcinoma of the ovary.

    A 52 year old woman presented with a five month history of emotional changes, voice changes, and of abdominal discomfort. Clinical, biochemical, and radiological examinations showed evidence of virilisation, raised testosterone, and a complex ovarian mass. microscopy of the resection specimen showed the tumour to be a transitional cell carcinoma of the ovary with luteinisation and hyperplasia of the intervening stromal cells. This is a unique case of virilisation caused by transitional cell carcinoma of the ovary. Theories as to the origin of testosterone production in non-functioning ovarian tumours are discussed.
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ranking = 1
keywords = lutein
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7/37. Recurrent severe hyperandrogenism during pregnancy: a case report.

    This report describes the case of a 28 year old woman with virilisation occurring in two successive pregnancies. Recurrent maternal virilisation is rare (seven previous reports) and this case is unique in its severity. Differential diagnoses include ovarian disease and fetal aromatase deficiency. New techniques to exclude a fetal cause were used in this case. This patient presented during the third trimester of her first pregnancy with rapid onset of hirsuitism, increased musculature, and deepening voice. A blood hormone profile revealed significant hyperandrogenism (testosterone, 72.4 nmol/litre; normal range, 0.5-3.0). She delivered a normal boy and maternal androgen concentrations returned rapidly to normal (testosterone, 0.8 nmol/litre). She presented two years later, during her second pregnancy, with similar symptoms and biochemistry (testosterone, 47.5 nmol/litre). Again, she delivered a healthy normal boy and androgens returned immediately to normal (serum testosterone, 2.0 nmol/litre). ultrasonography revealed no evidence of ovarian (or adrenal) masses in either pregnancy. umbilical cord venous blood sampling and placental assays revealed no evidence of fetal aromatase deficiency. Recurrent hyperandrogenism during pregnancy is rare. Ovarian luteoma rarely recurs and hyperreactio luteinalis does not lead to such pronounced androgen concentrations. Therefore, this patient has a unique ovarian condition that could be harmful to offspring and mother.
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ranking = 1
keywords = lutein
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8/37. Hyperreactio luteinalis presenting with marked hyperglycemia and bilateral multicystic adnexal masses at 21 weeks gestation.

    We present an unusual case in which a 23-year-old nulliparous patient was noted to have bilateral multicystic adnexal masses at 21 weeks gestation. Subsequent severe nausea, vomiting, and hyperglycemia with a serum glucose level of 450 mg/dL led to the diagnosis of gestational diabetes. Gradual development of marked maternal virilization ensued and was associated with a serum testosterone level of 4030 ng/mL (free testosterone level of 224 pg/mL), with normal serum dehydroepiandrostendione, estriol, and cortisol levels, leading to the diagnosis of hyperreactio luteinalis. Following vaginal delivery at 38 weeks gestation, gradual and complete spontaneous resolution of signs, symptoms, adnexal masses, diabetes, and testosterone levels were documented.
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ranking = 5
keywords = lutein
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9/37. A case of virilizing brenner tumor in a postmenopausal woman with stromal androgenic activity.

    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.
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ranking = 3
keywords = lutein
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10/37. Benign symptomatic hyperandrogenism in a postmenopausal woman.

    Hyperandrogenemia in postmenopausal women requires an evaluation to exclude pathologic ovarian or adrenal causes. Our patient exhibited no signs of hypercortisolism, congenital adrenal hyperplasia, or adrenal or ovarian neoplasia based on biochemical testing and pelvic sonography. We hypothesized that unexplained androgen excess in our patient was due to the development of gonadotropin-dependent excess ovarian stromal androgen production. This syndrome may be comparable to gestational hyperreactio luteinalis where elevated gonadotropins stimulate ovarian stromal androgen production. If tumor can confidently be excluded, such women may benefit from gonadotropin suppression with long-acting GnRH-a.
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ranking = 1
keywords = lutein
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