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PubMed2004

MRI of female genital and pelvic organs during sexual arousal.

We utilized contrast enhanced magnetic resonance imaging (MRI) to delineate the anatomy of the female genital and pelvic organs during sexual arousal. Eleven healthy pre-menopausal women and eight healthy post-menopausal women underwent MRI of the pelvis while watching an erotic video. A 1.5 Tesla MR system was used to produce T1-weighted images following administration of MS-325, a gadolinium-based blood pool contrast agent. Selected structural dimensions and enhancement were measured prior to and during sexual arousal. In both pre- and post-menopausal subjects, vestibular bulb and labia minora width increased with arousal. Enhancement measurements increased in the bulb, labia minora and clitoris in both pre- and post-menopausal subjects, and in the vagina in pre-menopausal subjects. There were no marked changes in size or enhancement of the labia majora, urethra, cervix, or rectum during sexual arousal in pre- or post-menopausal subjects. Using MRI, we observed specific changes in the female genitalia and pelvic organs with sexual arousal, in both pre- and post-menopausal women. MRI can potentially provide detailed anatomical information in the assessment of female sexual function, particularly with regard to changes in blood flow.

Adult↗
PubMed2004

Modern liberalism, female circumcision, and the rationality of traditions.

Tolerance is at the heart of Western liberalism, permitting mutually exclusive ideas and practices to coexist peacefully with one another, without the proponents of the differing ideas and practices killing one another. Yet, nothing challenges tolerance like the practice of sunna, female circumcision, clitorectomy, or genital mutilation. In this essay, I critique the Western critics of the practices, not in order to defend these practices, but rather to show that Western liberalism itself does not offer transcultural and transtemporal principles, for these principles only cohere within the tradition of liberalism. The Western critique of sunna often maps onto the bodies of African women Western symbolic notions of sexual and political freedom, as symbolized in the clitoris. The practices of sunna cohere within the web of beliefs, the tradition, of those who practice them and, thus, are rationally justified within those traditions. I offer Alasdair MacIntyre's notion of moral inquiry as a guide through the complex world of cultural and moral dialogue between differing traditions.

Circumcision, Female↗
PubMed2005

Intraoperative venous blood sampling to localize a small androgen-producing ovarian tumor.

BACKGROUND: Among other causes of virilization, ovarian tumors can be the cause of excessive androgen production. We report the case of a Leydig cell tumor of the ovary where diagnostic attempts to localize the source of hyperandrogenism preoperatively failed owing to relatively small tumor size. CASE: A 36-year-old woman presented with clinical signs of severe virilization including progressive balding, increased hirsutism, secondary amenorrhea and enlargement of the clitoris. Extensive work-up included endocrinological tests, pelvic ultrasound, magnetic resonance imaging, chromosomal analysis, norcholesterol scintigraphy and selective venous sampling, without direct localization of the source of hyperandrogenism. Persistently high plasma testosterone prompted an explorative laparotomy. Intraoperative selective blood sampling of the ovarian veins and palpation gave evidence of a right ovarian tumor, which was then removed. Histological examination revealed the presence of a pure Leydig cell tumor. CONCLUSION: Exploratory laparotomy with intraoperative selective blood sampling of the ovarian veins might be a useful approach in patients without accurate preoperative localization of androgen-producing tumors of the ovaries.

Adrenocorticotropic Hormone↗
PubMed2004

Granulocytic sarcoma of the ovotestis: an association of myelodysplastic syndrome and hermaphroditism.

Granulocytic sarcomas are extramedullary tumors (EMD) of malignant myeloid precursor cells. EMD or granulocytic sarcoma of ovary is rare disease. A 15-year-old girl had complaints of abdominal pain and weight loss for 3 months. On physical examination, there were hepatosplenomegaly and a painless mass under the umbilicus. Breast development was grade II. There was no clitoris hypertrophy. Her labia majora were separate and vagina hypoplastic. Hemoglobin level was 9.3 g/dl, white blood cells count 2.8 x 10(6)/1, platelet count 31.6 x 10(9)/1. There were dysplastic features in the blood and bone marrow cells. There were 10 and 22% blasts in the peripheral blood smear and bone marrow, respectively. The levels of serum follicle stimulating and luteinizing hormones were high. An inguinal mass (diameter 9.5 x 7.6) cm was detected on computed tomography. The histopathological diagnosis of this was obtained from laporascopy was composed of ovotestis and there was marked blastic infiltration in this ovotestis which had myeloid markers on flow cytometry. In the immunohistochemical analyses of ovotestis and bone marrow, blasts were positive for LCA, CD-13, CD-33 and CD 68. The cytogenetic analysis of the bone marrow shaved 46 XY karyotype. No response was achieved with combination chemotherapy and the patient died from progressive leukemia. Here we report a rare patient with myelodysplastic syndrome, EMD and hermaphroditism. To our knowledge this is the first case of MDS, EMD and hermaphroditism.

Adolescent↗
PubMed1987

Pancreatic exocrine aplasia, clinical features of leprechaunism, and abnormal gonadotropin regulation.

A female infant displaying morphological and clinical abnormalities is described. She had severe developmental delay, intrauterine growth retardation, and failure to thrive. Her features included an elfin facies, body and facial hirsutism, buttonlike nipples and prominent clitoris and labia. Endocrinological evaluation revealed markedly elevated serum gonadotropin levels. An LHRH stimulation test showed no LH response to LHRH but a rapid and pronounced FSH response. The patient died at 13 months following a respiratory infection. Postmortem examination revealed normal pituitary histology but an unusual pancreatic histology characterized by total absence of acinar cells. This patient had features suggestive of several clinical syndromes including leprechaunism, Shwachman syndrome, and Johanson-Blizzard syndrome, none of which, was completely satisfied. It is concluded that the infant may represent an atypical case of leprechaunism or a previously undescribed syndrome.

Facial Expression↗
PubMed2005

Lack of the SOX9 gene polymorphism in sex reversal dogs (78,XX; SRY negative).

The molecular background of the most frequent intersexuality syndrome in dogs (female-to-male sex reversal with the female karyotype and a lack of the SRY gene) is unknown. In this article, new cases of this syndrome are described in two unrelated American Staffordshire terrier dogs and one miniature pinscher dog subjected to cytogenetic and molecular analysis due to the presence of an enlarged clitoris. One dog was operated on and histological studies of the gonads revealed a testicular structure without signs of spermatogenesis, but the uterus wall appeared to be normal. All three dogs had female chromosome complements and lacked the Y-linked genes SRY and ZFY. Eight fragments, representing the vast majority of the coding sequence of the SOX9 gene, and two fragments of the 5' flanking region of this gene were analyzed. The studied fragments had identical DNA sequences when comparing the intersexual dogs with GenBank sequences (AY237827; NW139883). Thus a mutation in the coding sequence as well as the promoter region of the SOX9 gene might be excluded as a cause of this type of intersexuality. The importance of further studies of the 5' flanking region of this gene is discussed.

5' Flanking Region↗
PubMed1994

Infantile hemangiopericytoma versus infantile myofibromatosis. Study of a series suggesting a continuous spectrum of infantile myofibroblastic lesions.

The clinicopathologic features of 11 tumors, originally diagnosed as infantile hemangiopericytomas and with a spectrum of morphologic findings, are described. The age of the patients ranged from 6 days to 7 years; seven patients were younger than 1 year (mean, 2.25 years; median, 10 months); six were boys and five were girls. Three neoplasms were situated in skin or subcutis and seven in deep soft tissue; in one case the depth was unstated. Seven lesions arose in the lower limbs, and one each in the lumbar region, clitoris, chest wall, and soft tissue of the zygomatic region. One patient later was found to have two additional dermal tumors, one each on the anterior abdominal wall and the chest wall. Follow-up information in eight patients revealed local recurrence 12 years later in one case only. Histologically, all tumors showed distinctive features of infantile hemangiopericytoma, including immature cytology, multilobulated growth pattern, focal necrosis, and mitotic activity in varying degrees. Vascular invasion was noted in seven cases. Additionally, a second tumor cell component, composed of spindle-shaped myofibroblastic cells forming fascicles and micronodules, was evident at least focally. Both the spindle cells and more primitive round cells were positive for alpha-smooth muscle actin. Both cellular components showed a haphazard zoning arrangement. We discuss the clinicopathologic similarities between infantile hemangiopericytoma and infantile myofibromatosis and point out the differences between infantile and adult hemangiopericytoma. Our study suggests that there exists a broad spectrum of benign infantile myofibroblastic lesions containing an immature-appearing cellular component with a distinctive, hemangiopericytoma-like vascular pattern. Infantile myofibromatosis and so-called infantile hemangiopericytoma almost certainly represent different stages of maturation of the same (single) entity.

Child↗
PubMed1977

The child with ambiguous genitalia: a neonatal surgical emergency.

Although many birth defects pose a real threat to life if left uncorrected, the problem of ambiguous genitalia, if poorly or incorrectly handled, leads to a lifetime of unhappiness for both parent and child. The evaluation of these defects must be attended to with the same dispatch as any life threatening anomaly. A total of 18 children with ambiguous genitalia have been evaluated and treated in the past two years including nine females with virilizing adreno-genital syndrome, three infants with mixed gonadal dysgenesis, two infants with dysgenetic male pseudohermaphroditism, two males with severe perineal hypospadias, bifid scrotum, and undescended testicles, and two females with cloacal deformities. Precise diagnosis and most importantly proper gender assignments were made in 11 neonates by cytogenetic, biochemical, and miniaturized endoscopic techniques. Subsequent surgical reconstruction is governed by existing anatomy, not genetic sex, and has been completed in six children two to two and half years of age. Most are reconstructed as females with recession of the enlarged clitoris and vaginoplasty. Males with hypospadias are handled by standard techniques. Seven previously evaluated patients had been lost to follow-up for up to 13 years and were recently reconstructed. Surgical treatment in five older children with extreme virilization and moderate to severe gender confusion was technically successful but associated with pronounced emotional difficulties. Worse than delay in gender assignment, is making the wrong gender assignment, or failing to follow through with the original plan at an early age. Sample cases from each of these categories are presented in detail to illustrate the innovations in the diagnostic and therapeituc management of these children.

Adolescent↗
PubMed1996

Clinicopathologic features of vulvar squamous cell carcinomas exhibiting prominent fibromyxoid stromal response.

The frequency, distribution, and significance of a prominent fibromyxoid stromal response to tumor was examined in 51 consecutive cases of invasive squamous carcinoma of the vulva in which the lesion was totally excised and follow-up information available. The stromal response consisted of an admixture of myxoid change and immature collagen with fibroblasts at the tumor-stromal junction and was focally (< 25% of tumor) in 11 of the 51 cases (21%), regionally (26-50% of tumor) in seven cases (14%), diffusely (> 50% of tumor) in eight cases (16%), and not at all in 25 cases (49%). Tumors showing a prominent fibromyxoid stromal response (> 25% of the tumor) were typically flat or elevated ulcerative lesions, whereas carcinomas without a prominent fibromyxoid stromal response were more commonly exophytic. Sixty percent of the tumors with a prominent fibromyxoid stromal response involved the clitoris, compared with clitoral involvement in only 14% of carcinomas without a prominent response. Case showing prominent fibromyxoid stromal response were associated with a significantly older age group, poorer survival rate, and more extensive lymph node metastases than when fibromyxoid stromal response was not prominent. This behavior was manifested despite the fact that these tumors were not larger, more deeply invading, of higher grade, or more likely to show infiltrating patterns of invasion than when fibromyxoid stromal response was not prominent. Because the ability of a tumor to invade is believed to be related in a large part to changes in the relationship of tumor to stroma, a correlation between fibromyxoid stromal response and aggressive tumor behavior would suggest that a specific pattern of alteration in the interaction of tumor cells with stroma occurs in a subset of vulvar squamous carcinomas. The current findings may reflect the diverse etiology of vulvar carcinoma. If the characteristic features of vulvar carcinomas showing a prominent fibromyxoid stromal reaction as shown in this study can be confirmed on larger study populations, "squamous cell carcinomas with prominent fibromyxoid stromal reaction" may be a useful designation for these tumors.

Adult↗
PubMed1979

46, XY gonadal dysgenesis with secondary amenorrhea, virilization, and bilateral gonadoblastomas.

A phenotypic girl with secondary amenorrhea, enlargement of the clitoris, XY gonadal dysgenesis, and bilateral gonadoblastomas is described. The presence of secondary amenorrhea does not obviate the existence of a Y chromosome. The presence of the Y chromosome should be a warning that a gonadal tumor may be present and, therefore, gonadectomy must be done as soon as possible and preferably before puberty.

Adolescent↗
PubMed1999

Engineering tissues and organs.

Genitourinary tissues can be engineered in-vitro and in-vivo for reconstruction using selective cell transplantation in combination with acellular matrices. This technology involves an interdisciplinary approach combining techniques of cell biology and materials sciences towards the development of functional tissues or organs. Tissues and organs in urology, such as the bladder, clitoris, corpus cavermosum, kidney, testis, ureter and urethra have been created in the laboratory, with varying degrees of functionality. Cells have also been recently used in patients as bulking agents for the treatment of vesicoureteral reflux and urinary incontinence. As the science of tissue engineering evolves, one can expect a wider application of this technology to the armamentarium of urologic surgery.

Animals↗
PubMed2006

Defibulation to treat female genital cutting: effect on symptoms and sexual function.

OBJECTIVE: Women who have undergone type III female genital cutting may suffer long-term complications. Defibulation (reconstructive surgery of the infibulated scar) can alleviate some of these complications. We studied the physical and sexual outcomes after defibulation and evaluated both patient and husband satisfaction. METHODS: The medical records of 40 consecutive women with a history of type III female genital cutting who underwent defibulation between 1995 and 2003 were reviewed. Data collected included demographics, indications for the procedure, closure type, intraoperative and postoperative complications. Telephone surveys were conducted between 6 months and 2 years postprocedure to evaluate the long-term health and sexual satisfaction outcomes. RESULTS: Of 40 women identified as having undergone defibulation, 95% were Somali, 65% were married, and 73% were between the ages of 19 and 30. Primary indications for defibulation were being pregnant (30%), dysmenorrhea (30%), apareunia (20%), and dyspareunia (15%). Secondary indications were apareunia (20%), difficulty urinating (12.5%), and dyspareunia (10%). Sixty-five percent had a subcuticular repair. Forty-eight percent had an intact clitoris buried beneath the scar. None had intraoperative or postoperative complications. Of the 32 patients reached by telephone, 94% stated they would highly recommend it to others. One hundred percent of patients and their husbands were satisfied with the results, felt their appearance had improved, and were sexually satisfied. CONCLUSION: Defibulation is recommended for all infibulated women who suffer long-term complications. The complication rates are minimal, with high satisfaction rates among patients and their husbands. LEVEL OF EVIDENCE: II-3.

Adult↗
PubMed2005

Transobturator vaginal tape inside out for the surgical treatment of female stress urinary incontinence: anatomical considerations.

PURPOSE: We have recently described a novel surgical technique for female stress urinary incontinence, that is the transobturator vaginal tape inside out, which uses specific instruments for the passage of a synthetic tape from beneath the urethra toward the thigh folds. Herein we report the results of cadaver dissection performed to determine the anatomical trajectory of the tape and its relationships with neighboring neurovascular structures and organs. MATERIALS AND METHODS: Insertion of the transobturator vaginal tape inside out tape was performed by different surgeons in 12 freshly frozen female cadavers according to the standard procedure. The thigh, obturator, perineal and pelvic regions were dissected and tape trajectory was recorded. An additional cadaver was dissected without prior tape placement. RESULTS: The tape was inserted according to a certain consistent path, that is penetration from the suburethral space into a strictly perineal region limited medial and cranial by the levator ani muscle, caudal by the perineal membrane and lateral by the obturator internus muscle. This region corresponded to the most anterior recess of the ischiorectal fossa. The tape then perforated the obturator membrane and muscles, and exited through the skin after traversing adductor muscles and subcutaneous tissue. The tape was coursed away from 1) the dorsal nerve to the clitoris located more superficially below the perineal membrane, 2) the obturator nerve and vessels, and 3) the saphenous and femoral vessels. CONCLUSIONS: These findings strongly suggest that our transobturator technique is highly accurate, reproducible and safe, and it does not require perioperative cystoscopy.

Aged↗
PubMed2005

Lymphomas of the female genital tract: a study of 186 cases and review of the literature.

Malignant lymphomas in the female genital tract are rare, and those arising from this tissue system are extremely uncommon. Most pertinent reports lack clear references to the accepted classifications or failed to apply immunomarkers and molecular techniques for a reliable diagnosis. We analyzed a large group of patients with primary and secondary lymphomas of the female genital tract classified on the basis of the recent WHO consensus. A total of 186 patients with malignant lymphoma detected in the female genital tract were selected from the files of the Kiel Lymphoma Registry covering the period of 1974 to 2004. Stringent criteria were applied to separate systemic versus secondary lymphomas. All cases were reviewed on the basis of conventionally stained sections, relevant immunohistochemistry using the alkaline phosphatase/anti-alkaline phosphatase technique, and clinical information, as far as available. When required, gene rearrangement analysis was performed, including TCR-gamma chain gene and the three FR fragments of the IgG heavy chain gene. In addition, typical chromosomal translocations were detected by means of the FISH technique to verify the diagnosis, where needed. Thirty-seven percent of the cases were systemic lymphomas and 63% were mostly extranodal lymphomas primary to the female genital tract. The adnexa were involved in 87 cases, followed by uterine corpus in 23 cases, uterine cervix in 17 cases, portio in 9 cases, vagina in 11 cases, and vulva including clitoris in 8 cases. In 31 cases, two or more adjacent sites were involved. In both (primary and secondary) groups, the adnexa were the prevailing site of involvement. As expected, the overwhelming majority of cases were of B phenotype. The most frequent type of lymphoma proved to be diffuse large B-cell lymphoma, closely followed by follicular lymphoma, including all 3 grades of malignancy. Burkitt lymphoma showed a rather similar frequency. Marginal zone lymphoma occurred exclusively as primary lesions in the uterine mucosa. Lymphoplasmacytic lymphoma was restricted to the vulvo-vaginal area and occurred in women over 60 years of age. In conclusion, our study provides a thorough overview of various types of lymphoma affecting the female genital tract primarily or secondarily, which were classified on the basis of a widely accepted WHO classification. Although quite rare, our report should remind the pathologist of considering malignant lymphomas while reading biopsies taken from female genital organ.

Adolescent↗
PubMedDate not supplied

Vulvar Paget's disease with underlying adenocarcinoma simulating breast carcinoma: case report and review of the literature.

We report a case of extramammary Paget's disease with underlying adenocarcinoma simulating breast carcinoma of the vulva. An 82-year-old woman was found to have a 5 x 3-cm bulky tumor located in the left labium major, infiltrating to the clitoris, left labium minor, and left lateral tissue of the vulva. Small biopsy of the vulva showed intraepidermal proliferation of Paget cells. The patient underwent wide local excision of the vulvar tumor and dissection of left inguinal lymph nodes. Histopathological examination of the resected specimens revealed that Paget cells were distributed singly or tended to form small nests in the epidermis, and that association of these cells with the underlying carcinoma invading to the subcutis could be seen. The underlying carcinoma was composed of squamoid solid nests with central necrotic debris, mimicking 'comedocarcinoma' of the breast. In other areas, the tumor cells were present in tubular formations and solid cords reminiscent of invasive ductal carcinoma of the breast. Immunohistochemically, the Paget cells and the underlying carcinoma cells were positive for carcinoembryonic antigen, epithelial membrane antigen, estrogen receptors, and glandular keratins except for CK 20. We speculate that our case is vulvar Paget's disease presenting as a manifestation of underlying breast carcinoma of the vulva, which might have arisen from either the ectopic breast tissue or anogenital mammary-like glands.

Adenocarcinoma↗
PubMed1994

Inhibition of mammary tumors by pretreatment with 17 beta-estradiol in F344 rats induced with N-methyl-N-nitrosourea.

The influence of 17 beta-estradiol (E2) and prolactin was studied on N-methyl-N-nitrosourea (MNU)-induced mammary carcinomas (MCAs) in rats. MNU was intravenously injected once into seven-week-old female. female F344 rats at a dose of 50 mg/kg body weight. Groups of rats also received either 2.5 mg of E2 or a continuous supply of prolactin and/or growth hormone via transplanted MtT/F84 (mammo-somatotropic pituitary tumor). Rats were observed for up to 36 weeks after MNU administration. Although simultaneous administration of MNU and E2 did not much affect the occurrence of MCAs as compared to administration of MNU alone, rats treated with 2.5 mg of E2 for two weeks before MNU administration had significantly reduced occurrence of MCAs compared to those given MNU alone. In contrast, rats with MNU plus MtT/F84 showed high incidence and shortened latency of MCAs and they also had a high incidence of clitorial gland hyperplasias. Average pituitary weights and serum prolactin levels in E2-treated rats were greatly increased compared to those of MNU-alone rats. Average serum E2 levels were about 100 ng/ml in E2-treated rats and 0.05 ng/ml in rats without E2 treatment. Serum prolactin levels were greatly increased in rats with MtT/F84. The results indicated that pretreatment with E2 before MNU administration was inhibitory while increased prolactin caused by grafting MtT/F84 after MNU injection was promotive for the occurrence of MCAs in female F344 rats.

Adenocarcinoma↗
PubMed1981

Interstitial deletion of the short arm of chromosome 7 without craniosynostosis.

Two female infants with apparently identical interstitial deletions at bands p13 to p15 of chromosome 7 are presented. They differ in phenotype. The first infant has failure to thrive, retardation in development, normal head circumference with ridged metopic suture, blepharophimosis, epicanthal folds, mild hypotelorism, small low-set ears, and a bifid right toe. The second infant has a normal weight, length, and head circumference, blepharophimosis, epicanthal folds, widely spaced nipples, enlarged clitoris, and very large hands and feet. The two patients' clinical and karyotypic findings are compared with previous reports of structural abnormalities of the short arm of chromosome 7. Of the three cases in the literature, craniosynostosis was present in the two patients with deletion of band 7p14. Our observations, thus, suggest that deletion of bands 7p13 to 7p15, in contrast to more distal deletions at band 7p2, is not associated with craniosynostosis.

Abnormalities, Multiple↗
PubMed1993

Kaufman oculocerebrofacial syndrome: report of two new cases and further delineation.

Two unrelated Mexican girls, aged 14 months and 6 years respectively, with Kaufman oculocerebrofacial syndrome, are reported. Both showed psychomotor retardation, microcephaly, blepharophimosis and delayed growth as the main features; the infant also presented preauricular tags and large clitoris. Comparative analysis with previous cases reveals a heterogeneous syndrome in which the micro-brachycephaly, the mongoloid slanted eyes with different anomalies, the micrognathia and the neonatal respiratory distress are the most typical characteristics of this mental retardation syndrome.

Abnormalities, Multiple↗