Laparoscopic Excision of a Uterine Adenomatoid Tumor and a Coexisting Ovarian Teratoma: A Case Report and Literature Review.

Adenomatoid tumors (ATs) are rare, benign neoplasms occurring mainly in reproductive organs such as the uterus, ovaries, fallopian tubes, and testes. Uterine adenomatoid tumors (UATs) are generally incidentally diagnosed during histopathological examination of excisional biopsies performed for other indications, most commonly uterine leiomyomas. We herein present a 38-year-old woman who underwent laparoscopic excision of a uterine leiomyoma and a right ovarian teratoma. Microscopic examination of the excisional biopsy revealed that the enucleated uterine tumor was composed of proliferating glandular tissue covered with single-layered cells that were surrounded by proliferating smooth muscle cells, corresponding exactly to the features of UATs. The excised ovarian cyst was confirmed to be a typical mature cystic teratoma. According to these histopathological findings, the patient was finally diagnosed with a UAT and coexisting teratoma. No recurrence was detected up to 6 months after excision. To the best of our knowledge, this is the eighth case report on laparoscopically enucleated UATs. Although recurrence risk may be low in UATs, further case reports are necessary to elucidate the safety and validity of laparoscopic excision for UATs.

Primary alveolar soft part sarcoma of uterine corpus: a case report with immunohistochemical, ultrastructural study and review of literature.

BACKGROUND: Alveolar soft part sarcoma (ASPS) is a rare mesenchymal malignancy. ASPS usually occurs most commonly in the deep soft tissues of the thigh and buttock or the head and neck regions. ASPS that originate from the uterine corpus are even more rare, with only 10 previous cases reported in the English literature. CASE PRESENTATION: In our case, the alveolar features were completely lost and the tumour shows a solid, non-alveolar pattern and the nuclei have marked variation in nuclear size, and multinucleation. The correct pathological diagnosis has been made by immuno- histochemical and ultrastructural features, which rvealed overexpression of TFE3 and peculiar cytoplasmic crystalline inclusions. In this paper, an additional case of primary ASPS of uterine corpus is reported with immunohistochemical, ultrastructural study and review of literature in the effort to delineate its clinical and pathological features. In this unusual site, the diagnosis can be problematic because ASPS can mimic other primary or metastatic uterine neoplasms. CONCLUSIONS: Thus, in this unusual presentation an essential diagnostic marker is the nuclear over-expression of TFE3 as well as ultrastructural study, which reveals the presence of peculiar cytoplasmic crystalline inclusions.

Oncocytic adrenocortical neoplasm arising from adrenal rest in the broad ligament of the uterus.

Oncocytic adrenocortical neoplasm is characterized by abundant eosinophilic cytoplasm containing mitochondria, occasional nuclear atypia and diffuse growth pattern. Oncocytic adrenocortical neoplasm arising in adrenal rest is, however, extremely rare. We report a case of oncocytic adrenocortical neoplasm arising in adrenal rest of the broad ligament with associated marked lipomatous metaplasia. A well circumscribed tumor was accidentally detected in the pelvic cavity of a 29 year old Japanese woman, adjacent to the broad ligament of the uterus. The tumor was composed of large eosinophilic cells associated with diffuse growth pattern and abundant mature adipose tissue admixed with foci of clear cells. Both steroidgenic factor 1 (SF-1) and alpha-inhibin were immunohistochemically positive in tumor cells. Abundant mitochondria detected by immunohistochemical and electron microscopic examination confirmed the diagnosis of oncocytic adrenocortical neoplasm. The absence of necrosis, capsular and vascular invasion as well as the low mitotic index indicated the benign potential of this tumor. The tumor cells were also positive for dehydroepiandrosteron-sulfotransferase (DHEA-ST), 17ß-hydroxysteroid dehydrogenase type 5 (17ß-HSD5), 3ß-hydroxysteroid dehydrogenase (3ß-HSD) and steroid 17α-hydroxylase (P450-c17), suggesting a possible production of testosterone of this tumor. This is the first reported case of oncocytic adrenocortical adenoma arising in adrenal rest of the broad ligament.

Clinical and pathological characteristics, pathological reevaluation and recurrence patterns of cellular leiomyomas: a retrospective study in 76 patients.

OBJECTIVES: To analyze clinical and pathologic features as well as recurrence patterns of cellular leiomyomas (CL) in women who underwent surgical therapy for symptomatic disease. STUDY DESIGN: This retrospective study was conducted at the Department of Obstetrics and Gynecology, University Women's Clinic, Tuebingen, Germany. We identified all women who had CL on final diagnosis after surgery between January 1, 2000, and December 31, 2010. RESULTS: Our study sample comprised 76 women with a diagnosis of CL. A single uterine mass was present in 51.3% of the cases; in uteri with both CL and uterine leiomyomas (UL), the CL constituted the largest uterine mass in 20 of 21 (95.2%) cases. Additionally, in 98% of the uteri, CL were either the largest or the only uterine mass. Five women (6.6%; 5/76) had reported surgical procedures for symptomatic leiomyoma before the index surgery in our analysis. Three women underwent hysteroscopic resection of the leiomyomas and 2 women underwent abdominal myomectomy. Mean time to recurrence was 14.0 months (median 6.0; range, 4.0-52.0). Over the follow-up period, 6 women who underwent uterus-conserving surgery (12.0%; 6/50) with CL had leiomyoma recurrence. Five women underwent abdominal myomectomy and one underwent hysteroscopic resection of the CL. One patient had recurrence of a CL 43 months after abdominal myomectomy and underwent vaginal hysterectomy; the other five women had recurrences of UL. Mean time to recurrence was 28.6 months (median 12.5; range, 4.0-83.0). CONCLUSIONS: Recurrence rates of CL in our study group resemble recurrence rates of UL.

Quality of corrosion specimens prepared from material obtained during autopsies - a preliminary study.

AIM: Aim of this study was to assess the quality of the corrosion specimens obtained during autopsies of human body for scanning electron microscopy procedures. MATERIALS AND METHOD: Ninety seven uteri were obtained upon autopsy of women aged 25-56 years, deceased due to causes not related to disorders of the reproductive system. Fourty three of them contained large subserosal uterine leiomyomata. twenty uteri were injected with acrylic emulsion Liquitex R via the arteries or veins. Five of these uteri were next dissected and cut into slides on a microtom. the remaining uteri were injected with 60-80 ml of mercox CL-2r resin, next macerated and studied under scanning electron microscope (JEOL SEM 35-CF scanning electron microscope at 20-25 kV). RESULTS: Best human specimens were obtained from the autopsies carried out possibly early after the deceased, young aged (between 25 and 45) and died because of multitrauma not associated with the pelvic injury. CONCLUSIONS: Specimens obtained from autopsies can be used for scanning electron microscopy however under several conditions, specially the time between death and undertaking the injection procedures and the age of the individual, because of the process of artherosclerosis.

Vascular structure of outer myometrial uterine leiomyomata - a preliminary SEM and immunohistochemical study.

AIM: The main goal of this study was assessment of vascular structure of uterine leiomyomata localized between outer myometrium and endometrium. MATERIALS AND METHODS: The study was carried out on thirty two human uteri collected upon autopsy. Vessels were injected with synthetic resin, next corroded and coated with gold, finally observed using scanning electron microscope. Next ten uteri were injected with acrylic emulsion and studies using immunohistochemical staining for von Willebrandt's factor. RESULTS: Vascular structure of outer myometrial leiomyomata was quite similar to those observed in the middle of muscular layer of uterus, characterized by relatively dense 'vascular capsule', consisted of flattened vein, arterioles and capillaries. CONCLUSIONS: Structure of outer myometrial uterine leiomyomata was similar to those observed during growth within myometrium.

Blood vessels of the intratumoral septa in uterine leiomyomata.

The angioarchitecture of fibroid intratumoral septa was studied using 32 uteri obtained during necropsies of the females aged between 35-57. The whole vascular bed of 16 uteri was injected with synthetic resin Mercox CL-2R and then the uteri were corroded in potassium hydroxide. Next 16 uteri were injected with acrylic emulsion, Liquitex R. Their vascular bed was studied using immunohistochemistry for von Willebrandt's factor. Immunohistochemistry allowed to visualize the vessels within the intratumoral septa, while SEM allowed to differentiate the vessels, which were mainly the venules and the veins. Apart from the veins the intratumoral septa were consisted of small arteries and capillaries.

Gestrinone inhibits growth of human uterine leiomyoma may relate to activity regulation of ERα, Src and P38 MAPK.

The study was to investigate the effect of gestrinone on the growth of human uterine leiomyoma cells and on the levels and activity of p38, Src and estrogen receptor alpha (ERα). Human uterine leiomyoma cells were cultured and treated with dimethylsulfoxide (DMSO) or a gestrinone concentration gradient. Morphological changes were observed and apoptosis was evaluated. Levels of p38 and phosphorylated-p38 (pp38) were assayed by enzyme-linked immunosorbent assay (ELISA). Levels of ERα and Src were analyzed using real-time RT-PCR and Western blotting. The result showed that gestrinone significantly inhibited the growth of cultured human uterine leiomyoma cells in a concentration- and time-dependent manner, with a 50% inhibitory concentration (IC(50)) value and corresponding 95% confidence intervals (CI) of 43.67 (23.46∼81.32), 27.78 (12.51∼61.68) and 15.25 (7.17∼32.43) µmol/L at 20, 40 and 60h, respectively. Compared with control-treated leiomyoma cells, gestrinone significantly reduced both the expression of ERα (P<0.05) and the levels of phospho-Ser167-ERα (P<0.05). Gestrinone also markedly suppressed the level of phospho-Tyr416-Src (P<0.05). Moreover, gestrinone significantly increased the ratio of phospho-p38/p38 mitogen-activated protein kinase (MAPK) (P<0.05). However, no significant increase in apoptosis or cell cycle arrest was observed (P>0.05) in response to the tested concentrations of 0.1 to 3.0µmol/L. As a conclusion, gestrinone suppresses the proliferation of uterine leiomyoma cells mainly by regulating the activity of ERα/Src/p38 MAPK in a concentration-dependent manner at a low concentration of 0.1∼3.0µM, but not significantly regulating apoptosis. Gestrinone opposes the growth of uterine leiomyoma through multiple genes.

Uterine fibroid pseudocapsule studied by transmission electron microscopy.

OBJECTIVE: The fibroid pseudocapsule is a structure which surrounds the uterine fibroid, separates it from the uterine tissue and contains a vascular network rich in neurotransmitters like a neurovascular bundle. The authors examined the composition of the fibroid pseudocapsule using electron microscopy. STUDY DESIGN: Twenty non-pregnant patients were submitted to laparoscopic myomectomy by the intracapsular method and samples of the removed pseudocapsules were analyzed using transmission electron microscopy. RESULTS: At the ultrastructural level the pseudocapsule cells have the features of smooth muscle cells similar to the myometrium. So, the pseudocapsules are part of the myometrium which compresses the leiomyoma. CONCLUSION: This ultrastructural feature suggests that when removing fibroids their pseudocapsules should be preserved. This study confirms preliminary evidence that pseudocapsules contain neuropeptides together with their related fibers, as a neurovascular bundle. The surgeon's behavior should be directed to carefully control and spare this muscular surrounding tissue during fibroid excision, in order to preserve the myometrium as much as possible.

Pure alveolar rhabdomyosarcoma of the uterine corpus.

Herein is presented a very rare case of alveolar rhabdomyosarcoma in the uterine corpus of a 72-year-old woman. The wall of the uterine corpus was replaced by multiple whitish-yellow, friable nodules, measuring up to 6 cm. Microscopically, the tumor was predominantly composed of round to polygonal cells arranged in an alveolar, papillary or nest pattern intermingled with multinuclear giant cells with abundant eosinophilic cytoplasm. Extensive sampling failed to show epithelial elements. Immunohistochemically, the tumor was positive for striated muscle markers such as myoglobin, myoD1 and myogenin. Metastatic lesions were found in the retroperitoneum and pelvic lymph nodes. The patient was treated by postoperative chemotherapy, but she died of systemic metastases 12 months after surgery.

Uterine pleomorphic rhabdomyosarcoma: a case report and review of literature.

Primary pleomorphic rhabdomyosarcoma of the uterus is a very rare neoplasm. We describe a 65-year old female with this diagnosis, who underwent an abdominal hysterectomy with bilateral salpingo-oophorectomy and lymph node dissection. Also included is a discussion on the different types of rhabdomyosarcoma with reviews of their histological features, epidemiology and common sites of origin.

Ewing sarcoma-primitive neuroectodermal tumor of the uterus: a clinicopathologic, immunohistochemical and ultrastructural study of one case.

INTRODUCTION: Ewing sarcoma-primitive neuroectodermal tumors (ES/PNET) constitute a family of neoplasms characterized by a continuum of neuroectodermal differentiation. ES/PNET of the uterus is rare. There are 43 cases published in the English literature as far as we know. We describe an additional case. CASE REPORT: A 56-year-old woman presented with a 2-month history of irregular menopausal vaginal bleeding. After surgical excision, microscopic, immunohistochemical and electron microscopic examination suggested the diagnosis of ES/PNET. The patient underwent combined chemotherapy consisting of ifosfamide, etoposide, and cisplatin. She was alive with no evidence of recurrence or metastasis after 41 months of the initial operation. DISCUSSION: In spite of the rarity of ES/PNET, we should consider it in the differential diagnosis of small cell neoplasms of the uterus.

Mucinosis y vasculitis a nivel uterino asociadas a lupus eritematoso / Uterine mucinosis and vasculitis associated with lupus erythematous

Uterine mucinosis and vasculitis associated with lupus erythematous(AU)

The association of cutaneous papulonodular mucinosis with lupus erythematosus has long been recognized. Massive cutaneous deposits of mucin and periorbital mucinosis have also been reported in patients with lupus. Recently, a report was published of two cases of mucin deposits in the myometrium of patients with lupus, denominated "myxoidosis" by the authors. A case is presented of vasculitis and diffuse, predominately myometrial, mucinosis, in the uterus of a patient with lupus erythematosus. The term "mucinosis" and not "myxoidosis" is recommended, even when referring to the uterus, in order to unify the terminology of pathological findings that have similar histological and histochemical features to the cutaneous lesions found in lupus erythematosus(AU)

Morphologic patterns of human endometrial epithelium in women with uterine myomata treated with leuprorelin acetate.

AIM: We studied morphologic modifications of the endometrium induced by leuprorelin acetate, a gonadotropin-releasing hormone agonist, in women with uterine myomata. METHODS: Transmission and scanning electron microscopy observations were performed after 2 or 6 cycles of therapy (every 28 days). RESULTS: A near-normal endometrium was observed after 2 months of therapy, while treatment with 6 cycles of leuprorelin acetate induced a uniform morphologic regression of the uterine mucosa. CONCLUSIONS: The study demonstrates that leuprorelin acetate induces a unique and time-dependent regression of the endometrial mucous membrane.

Microscopía electrónica de barrido de la hiperplasia en la vellosidad molar / Scanning electron microscopy of placental hyperplasia

Objetivo: Estudio de la superficie externa del sincitiotrofoblasto de vesículas de mola hidatidiforme, fue rastreada utilizando la microscopía electrónica de barrido. Método: Especimenes de material molar de 21 semanas de embarazo se fjaron en 2 por ciento de glutaraldehido 0,1 M a 4ºC en sala de parto y posteriormente post fjados en 1 por ciento de tetraoxido de osmio siguiendo los procedimientos convencionales de la microscopía electrónica de barrido como deshidratación, desecado de punto crítico, cubrimiento iónico y observación con el microscopio electrónico de barrido. Resultados: Los resultados revelan cambios morfológicos en la membrana plasmática sincitial, desde superfcie aplanadas con pequeños gránulos o promontorios, hasta la formación de numerosas prolongaciones de membrana que originan pliegues, bandas o columnas, las cuales se ramifcan intensamente, tomando contacto entre sí para conformar una complicada trama, que deja un retículo de espacios como cavernas, las cuales se abren hacia el espacio intervelloso siendo la expresión de una intensa proliferación de membranas en hiperplasia. Conclusión: La observación de esta trama permite un mejor entendimiento de la estructura de la mola comparada con las imágenes de microscopía de luz.

Objective: Study of the external surface of the syncytiotrophoblast in vesicles of hydatidiform mole was examined and analysed using scanning electron microscopy. Method: Vesicles of molar material were taken at 21 weeks of pregnancy and fxed in 2 percent of glutaraldehyde 0.1 M at 4 °C in delivery room and furtherly post-fxed in 1 percent of tetroxide of osmium according with conventional procedures of the scanning electron microscopy as dehydration, critical point drying, surface coating and examination using scanning electron microscope. Result: The fndings reveal the morphological changes in the syncytial membrane from smooth surface with small bridges or granules to the formation of numerous prolongations of plasma membrane, which produce folds, bands or columns with ramifcations, that get in touch organizing a complex net that contain spaces opened to the intervillous space. This is an expression of the proliferation of syncytial plasma membrane during the hyperplasia. Conclusion: The tridimensional observation of this net complex permit a better understanding of the structure of the molar vesicle when these images are compared with those of light microscopy.

Chorangiocarcinoma: a case report and review of the literature.

Chorangiocarcinoma is the name designated to a chorangioma with trophoblastic proliferation manifesting increased proliferative activity. Only 3 such cases have been published so far. Other studies challenged this entity by demonstrating that proliferation of the trophoblast around chorangioma is a common phenomenon. We present a case of a unique vascular lesion in a term placenta with a malignant trophoblastic component. Microscopic examination of a well-demarcated placental mass revealed a chorangioma with multiple nodules composed of pleomorphic cells displaying focal multinucleation, large areas of necrosis, and high mitotic activity. Immunohistochemical stains of these cells were strongly positive for pancytokeratin and the beta subunit of human chorionic gonadotropin and focally positive for HSD3B1. There was no invasion of the basement membrane, and no free-floating tumor cells in the intervillous space. No evidence of metastasis was found on follow-up of the mother and newborn. It is concluded that the tumor presented herein, displaying a histologically unequivocal malignant trophoblastic component in a benign chorangioma, is a true chorangiocarcinoma, and should be included within the category of gestational neoplasia as a tumor closely related to choriocarcinoma.

A case of pure uterine lipoma: immunohistochemical and ultrastructural focus.

BACKGROUND: Pure uterine lipoma is a rare clinical event and only a few cases have been reported in literature. The histogenesis of these lesions is still debatable. Preoperative diagnosis is difficult and should be pathologically confirmed postoperatively. CASE: We report the case of a 58-year-old woman who presented with pelvic pain and postmenopausal uterine bleeding. The hysterectomy specimen showed a pure intramural lipoma of the uterus. An immunohistochemical study revealed that the lipomatous tissue was reactive to S-100, vimentin, actin and desmin. Electron microscopy examination revealed bundles of spindle cells with intracytoplasmatic vacuoles and parallel-arranged intermediate filaments in the surrounding zone, in which adipose cells were mixed with muscular cells. DISCUSSION: Clinical and histological diagnosis of pure uterine lipomas are described and a possible involvement of fatty metaplasia of smooth muscle cells in the development of pure uterine lipomas is discussed.