Cases reported "Amenorrhea"

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1/15. Hypergonadotropic ovarian failure associated with an inherited mutation of human bone morphogenetic protein-15 (BMP15) gene.

    Hypergonadotropic ovarian failure is a common cause of female infertility. It is a heterogeneous disorder that, in the most severe forms, is a result of ovarian dysgenesis (OD). Most OD cases are associated with major X-chromosome abnormalities, but the pathogenesis of this disorder is still largely undefined in patients with a normal karyotype. Animal models showed the important role in female reproduction played by the product of a gene located at Xp11.2 in humans (BMP15). BMP15 is an oocyte-specific growth/differentiation factor that stimulates folliculogenesis and granulosa cell (GC) growth. We report two sisters with a normal karyotype who are affected with hypergonadotropic ovarian failure due to OD. The familial presentation suggested a genetic origin, and candidate genes were screened for mutations. A heterozygous nonconservative substitution in the pro region of BMP15 (Y235C) was identified in both sisters but not in 210 control alleles. This mutation was inherited from the father. Mutant BMP15 appears to be processed abnormally, is associated with reduced GC growth, and antagonizes the stimulatory activity of wild-type protein on GC proliferation. In conclusion, the first natural mutation in human BMP15 is associated with familial OD, indicating that the action of BMP15 is required for the progression of human folliculogenesis. This condition represents an exceptional example of X-linked human disease exclusively affecting heterozygous females who inherited the genetic alteration from the unaffected father. BMP15 defects are involved in the pathogenesis of hypergonadotropic ovarian failure in humans.
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ranking = 1
keywords = granulosa cell, granulosa
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2/15. Inhibin-producing ovarian granulosa cell tumor as a cause of secondary amenorrhea: case report and review of the literature.

    We report the case of 31-year-old patient with an inhibin B-secreting granulosa cell tumor of the left ovary who presented with secondary amenorrhea. Preoperative serum hormonal levels were as follows: follicle-stimulating hormone (FSH) 0.3 mIU/mL, luteinizing hormone (LH) 9.81 mIU/mL, estradiol 142.0 pg/mL and inhibin B 2429 pg/mL. gonadotropin-releasing hormone (GnRH) test revealed no FSH response and a normal LH response. After removal of the tumor, the levels of FSH and inhibin B returned to within the normal range, and regular menses resumed 27 days postoperatively. In premenopausal women, secondary amenorrhea may be the initial manifestation of granulosa cell tumor. A low FSH level coupled with normal levels of E2 and LH, the inhibition of the FSH response to GnRH and an elevated inhibin level suggest the presence of an inhibin-secreting ovarian tumor and also rule out the possibility of isolated FSH deficiency.
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ranking = 6
keywords = granulosa cell, granulosa
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3/15. Androgenic adult granulosa cell tumor in a teenager: a case report and review of the literature.

    The clinicopathologic findings of the third case of androgenic adult granulosa cell tumor in patients younger than 15 years was presented and discussed in the light of the literature. A patient complaining of secondary amenorrhea and hirsutism with elevated levels of plasma total testosterone, dehydroepiandrosterone sulfate, free androgen index and serum inhibin A, and a left ovarian septated, cystic mass was admitted to the hospital. The inhibin A level was within normal levels in the first month postoperatively. Inhibin A could be a tumor marker of utmost importance particularly in patients with androgenic or hyperestrogenic symptoms, especially in cases where benign criteria are abundant such as young age, nonincreased levels of classic tumor markers, and ultrasonographic appearance without any suspicion of malignancy.
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ranking = 5
keywords = granulosa cell, granulosa
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4/15. Unusual presentation of juvenile granulosa cell tumor of the ovary.

    BACKGROUND: We present a case report of juvenile granulosa cell tumor of the ovary (JGCT) with an unusual clinical presentation and hormonal secretion. CASE: A 16-yr-old girl had developed spontaneous menarche at the age of 12 yr, but after this initial menstrual bleeding she had no further periods for 4 yr. She had no clinical signs of virilization. Endocrinological studies detected high levels of DHEA, 17 hydroxyprogesterone (17OH-P), insulin and PRL, an exaggerated DHEA response after ACTH stimulation, and low FSH and high LH values after GnRH. An ultrasound examination revealed an irregular structure and increased diameters of her right ovary, due to the presence of a cyst. Because exploratory laparoscopy revealed the presence of a right ovarian mass, her right ovary was removed. JGCT was diagnosed. Ten days after surgery, menstrual bleeding initiated. Endocrinological evaluation after the operation showed that 17OH-P, insulin and basal FSH and LH serum values had returned to normal, while DHEA levels had decreased to within the upper limit of the normal range. Only PRL levels remained unchanged. CONCLUSION: Our patient presented some unusual characteristics. She did not have precocious puberty, but secondary amenorrhea. Hormonal secretion consisted mainly of androgens, even though clinical signs of virilization were not present.
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ranking = 5
keywords = granulosa cell, granulosa
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5/15. Inhibin as a marker for granulosa-cell tumors.

    Inhibin is a peptide hormone normally produced by ovarian granulosa cells. It reaches a peak of 772 /- 38 U per liter in the follicular phase of the menstrual cycle and is undetectable in the serum of menopausal women. To determine whether measurements of serum inhibin levels would provide a biochemical marker of the presence or progression of ovarian granulosa-cell tumors and their metastases, we measured the serum immunoreactive inhibin concentrations in six women with such tumors. Three women had been treated by hysterectomy and bilateral salpingo-oophorectomy. In the two women with residual or recurrent disease, the serum inhibin levels were abnormally elevated 5 and 20 months before the clinical manifestations of recurrence became evident. The maximal concentrations approached 3000 U per liter. The serum inhibin level remained undetectable in one patient who was disease-free for 11 years. serum inhibin concentrations were also elevated in three women with amenorrhea and infertility that resulted from small granulosa-cell tumors. After the removal of the tumors, the serum inhibin levels in these women became normal, and fertility returned. There was a significant negative correlation between the serum concentrations of inhibin and follicle-stimulating hormone, in a manner consistent with the autonomous production of inhibin by granulosa-cell tumors. We conclude that granulosa-cell tumors produce inhibin. Since serum inhibin levels reflect the size of the tumor, measurements of inhibin can be used as a marker for primary as well as recurrent disease.
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ranking = 3.3600771680409
keywords = granulosa cell, granulosa
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6/15. Pregnancies after premature ovarian failure.

    Six women who conceived after a diagnosis of premature ovarian failure are discussed. Two pregnancies occurred while the women were receiving conjugated estrogen therapy, two while taking oral contraceptives, and two women conceived spontaneously. The possible role of exogenous estrogens in sensitizing the granulosa cells to the effect of follicle-stimulating hormone and thereby inducing ovulation and conception in some women with premature ovarian failure is examined.
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ranking = 1
keywords = granulosa cell, granulosa
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7/15. Lipid cell thecomas of the ovary.

    An ovarian tumour of mixed thecomatous and lipid cell structure is described and the clinical and pathological features of this and eleven previously reported cases are reviewed. Functionally the tumours resemble lipid cell tumours in being mainly androgenic and are similar in size range and age incidence to the 'adrenal' type. Three tumours however contained hilus (Leydig) cells with Reinke crystalloids. The tumours furnish additional evidence of the stromal origin of lipid cell tumours and the term 'lipid cell thecoma' is proposed for them as indicating their main morphological affinities. It is suggested that fibromas, fibrothecomas, thecomas, lipid cell thecomas and lipid cell tumours form a series of related stromatogenous tumours of relatively simple structure with functional correlations as one moves from left to right.
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ranking = 0.068670126422154
keywords = cell tumour
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8/15. Binovular follicles in the adult human ovary.

    The human ovary may contain binovular or polyovular follicles at birth, but they are unusual later in life. Binbovular follicles were found in bilateral ovarian biopsies from a patient with primary amenorrhea who had been treated with exogenous gonadotropins. The fine structural morphology of these follicles has shown that both oocytes were in the resting meiotic prophase, that their respective vitelline bodies faced each other across the intervening plasma membranes, and that the adjacent oocytes made mutual contacts similar to those found between granulosa cells and oocytes. From the appearance of the multivesicular bodies in nearby stromal fibroblasts it is suggested that these follicles had formed by fusion of two adjacent primordial follicles by a mechanism similar to the depolymerization of the ground substance in the apex of the Graafian follicle that leads to ovulation.
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ranking = 1
keywords = granulosa cell, granulosa
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9/15. Androgenic granulosa cell tumors of the ovary. A clinicopathologic analysis of 17 cases and review of the literature.

    We reviewed the clinicopathologic features of 17 androgenic granulosa cell tumors. The patients, two of whom were pregnant, ranged from 2 to 58 years of age (average age, 22.5 years). Fourteen of the patients were masculinized as evidenced by various combinations of facial acne, temporal recession of hair, clitorimegaly, deepening of the voice, and a male escutcheon. Five of these patients had amenorrhea; three of the patients demonstrated hirsutism only. The plasma testosterone level is known to have been elevated preoperatively in nine patients, including the three patients with hirsutism. Four prepubertal patients also demonstrated hyperestrinism as manifested by sexual pseudoprecocity. At laparotomy, all of the tumors were stage lai. Five of the tumors formed unilocular and two of the tumors formed multilocular thin-walled cysts that ranged from 23 to 36 cm in diameter. Nine of the remaining tumors were solid, and one tumor was solid and cystic; these tumors averaged 7 cm in diameter. Eleven tumors were granulosa cell tumors of the adult type, and six tumors were juvenile granulosa cell tumors. The only clinically malignant tumor in the series was a unilocular, cystic, adult granulosa cell tumor that recurred 18 months postoperatively and was fatal within four years.
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ranking = 8
keywords = granulosa cell, granulosa
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10/15. The gonadotrophin resistant ovary syndrome: a curable disease?

    A patient with the resistant ovary syndrome is reported. It is suggested that lack of synchronization between steroidogenesis in the granulosa and theca cells is responsible for the (acquired) occurrence of this syndrome. feedback inhibition of pituitary gonadotrophin secretion was achieved by exogenously administered ovarian steroid hormones. All protein and steroid hormone levels returned to normal and spontaneous ovulatory cycles returned after withdrawal of medication. It is concluded that the so called "resistant ovary syndrome" is not an irreversible process.
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ranking = 0.29500964600512
keywords = granulosa
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