Cervical Mesonephric Adenocarcinoma: A Case Report of a Rare Gynecological Tumor from Embryological Remains of the Female Genital Tract.

INTRODUCTION: Malignant mesonephric tumors are uncommon in the female genital tract, and they are usually located where embryonic remnants of Wolffian ducts are detected, such as the uterine cervix. The information about these tumors, their treatment protocol, and prognosis are scarce. CASE REPORT: A 60-year-old woman with postmenopausal vaginal bleeding was initially diagnosed with endometrial carcinoma. After suspicion co-testing, the patient underwent a loop electrosurgical excision of the cervix and was eventually diagnosed with mesonephric adenocarcinoma. She was subjected to a radical hysterectomy, which revealed International Federation of Gynecology and Obstetrics (FIGO) IB1 stage, and adjuvant radiotherapy. The follow-up showed no evidence of recurrence after 60 months. CONCLUSION: We present the case of a woman with cervical mesonephric adenocarcinoma. When compared with the literature, this case had the longest clinical follow-up without evidence of recurrence, which reinforces the concept that these tumors are associated with a favorable prognosis if managed according to the guidelines defined for the treatment of patients with cervical adenocarcinomas. Though a rare entity, it should be kept in mind as a differential diagnosis for other cervical cancers.

Mesonephric adenocarcinoma in the uterine corpus: A case report and review of the literature.

Mesonephric adenocarcinoma (MNA) is a rare malignancy arising from the mesonephric remnant of the female reproductive tract, typically found in the cervix. MNA is uncommon in the uterine corpus, only 33 cases have been described in the literature. A 55-year-old postmenopausal woman presented with pink vaginal discharge and bilateral hip pain for 2 months, with the help of histopathologic observation and immunohistochemical staining, a diagnosis of "MNA" was made. The tumor invaded the whole layer of myometrium without endometrium involvement, mesonephric remnants and hyperplasia of the mesonephric duct were also found at the periphery of the neoplasm. After the operation, the patient was treated with 3 cycles of chemotherapy. The patient was followed for 6 months with disease. Further experience to diagnose and cure this rare tumor is warranted.

A case of mesonephric adenocarcinoma of the vagina with a 1-year follow-up.

Mesonephric adenocarcinoma deriving from remnants of vaginal mesonephric ducts is one of the rarest tumors of the female genital tract with only three cases reported till date in international literature. Differential diagnosis from other aggressive tumors is complex and controversies exist in the literature regarding the biological behavior, prognosis, and optimal management strategies of these tumors. A 58-year-old woman presented with a large mass extending from the right adnexal region to the perineum and labia majora. CA125 was increased. A radical excision of the lesion with pelvic and para-aortic lymphadenectomy was performed. A well-capsulated mesonephric adenocarcinoma in a background of vaginal mesonephric remnants was diagnosed. Tumor cells showed immunoreactivity for pancytokeratin, cytokeratin (CK), CD 10, epithelial membrane antigen, vimentin, and calretinin; indeed they were negative for carcinoembryonic antigen, CK 20, estrogen receptor, and progesterone receptor. No evidence of lymph node involvement or metastatic disease was observed. The patient did not receive any adjuvant therapy and is alive and clinically free of disease at 1-year follow-up. In spite of the aggressive biological behavior attributed in literature to mesonephric carcinomas, which is probably due to the complex differential diagnosis with other müllerian tumors, the favorable course of our patient further supports the hypothesis that malignant mesonephric carcinomas may not behave aggressively and that radical surgery alone may be curative.

[Mesonephroid carcinoma of the urinary bladder (clear cell carcinoma). Two case reports and review of the literature in a rare variation of the primary adenocarcinoma]. / Das mesonephroide Karzinom der Harnblase (Klarzellkarzinom). Zwei Kasuistiken und Literaturübersicht über eine seltene Variante des primären Adenokarzinoms.

Mesonephroid adenocarcinoma of the bladder may be a malignant form of nephrogenic adenoma or nephroid metaplasia. The lesion is extremely rare in the urinary bladder, and to our knowledge 19 cases have been reported in the literature. We report two cases of mesonephroid adenocarcinoma of the bladder which were treated by radical cystectomy.

Cervical mesonephric adenocarcinoma: A case report of a rare gynecological tumor from embryological remains of the female genital tract / Adenocarcinoma mesonéfrico cervical: Relato de caso de um tumor ginecológico raro de restos embriológicos do trato genital feminino

Abstract Introduction Malignant mesonephric tumors are uncommon in the female genital tract, and they are usually located where embryonic remnants of Wolffian ducts are detected, such as the uterine cervix. The information about these tumors, their treatment protocol, and prognosis are scarce. Case report A 60-year-old woman with postmenopausal vaginal bleeding was initially diagnosed with endometrial carcinoma. After suspicion co-testing, the patient underwent a loop electrosurgical excision of the cervix and was eventually diagnosed with mesonephric adenocarcinoma. She was subjected to a radical hysterectomy, which revealed International Federation of Gynecology and Obstetrics (FIGO) IB1 stage, and adjuvant radiotherapy. The follow-up showed no evidence of recurrence after 60 months. Conclusion We present the case of a woman with cervical mesonephric adenocarcinoma. When compared with the literature, this case had the longest clinical follow-up without evidence of recurrence, which reinforces the concept that these tumors are associated with a favorable prognosis if managed according to the guidelines defined for the treatment of patients with cervical adenocarcinomas. Though a rare entity, it should be kept in mind as a differential diagnosis for other cervical cancers.

Successful treatment of pure endodermal sinus tumors in adult men.

Seven adult men with pure endodermal sinus tumors (EST) were treated with cyclical combination chemotherapy Cytoxan (cyclophosphamide; Bristol-Myers Company, Evansville, IL), Adriamycin (doxorubicin; Adria Laboratories, Columbus, OH), and cisplatin/vinblastine and bleomycin (CISCAII/VBIV) and surgery at the University of Texas M.D. Anderson Hospital and Tumor Institute at Houston from 1978 through 1985. Six tumors were of extragonadal origin (four anterior mediastinum, one pelvic, one prostate), and one was of gonadal origin with retroperitoneal metastasis. All patients presented with advanced local disease and a relative absence of distant metastasis. Alpha-fetoprotein (AFP) levels were elevated in six patients (median, 4,400 ng/mL; range, 2,580 to 31,200 ng/mL). Six patients achieved a complete remission (CR): one with chemotherapy alone, one with initial surgery followed by chemotherapy, and four with chemotherapy followed by consolidative surgery. The remaining patient died of progressive disease. Of the six patients who achieved a CR, five are alive with no evidence of disease (+17, +23, +34, +43, +59 months); one patient developed recurrent disease at 6 months after completion of therapy and is currently undergoing salvage chemotherapy. Of the four patients who underwent postchemotherapy surgery, three were operated on for a marker-negative stable mass; in these patients, no viable tumor was found at pathologic review. The remaining patient underwent surgery for a stable mass with a persistent elevation in AFP levels. He was found to have 95% necrosis with 5% viable tumor and remains disease free without further therapy. The observed changes in AFP levels correlated with regression and progression of tumor; a normal AFP was consistent with a CR, and elevation was consistent with residual tumor. These seven patients demonstrate that when adult men with EST are treated aggressively with combination chemotherapy and surgery, high cure rates can be achieved.

Mesonephric adenocarcinomas of the uterine cervix: a study of 11 cases with immunohistochemical findings.

Mesonephric adenocarcinoma is a rare variant of cervical carcinoma with relatively few, well-documented cases reported. We describe the clinicopathologic and immunohistochemical features of 11 examples of this neoplasm, which occurred in women between the ages of 35 and 72 years (mean, 52 years). Most (64%) patients had abnormal vaginal bleeding. Eight tumors were stage IB, and one each was stage IIB and IVB; in one, the stage was unknown. Microscopically, the carcinomas showed various morphologies, most commonly a small tubular pattern or a ductal pattern resembling endometrioid adenocarcinoma; one tumor had an associated malignant spindle cell component. Ten neoplasms were adjacent to hyperplastic mesonephric remnants. Follow-up in 10 cases showed six patients to be alive without evidence of recurrence after a mean of 4.8 years. The patients with stage IIB and IVB disease had local recurrences after 2.2 and 0.7 years and died of progressive disease at 3.2 and 0.8 years, respectively. In a patient with stage IB disease, a mediastinal metastasis and a malignant pleural effusion developed 5.6 years after diagnosis, and the patient died of disease at 6.2 years. Another patient with stage IB disease and a positive vaginal cuff margin that recurred locally after 1.7 years received chemotherapy and was alive and clinically free of disease at 2.5 years. Mesonephric adenocarcinomas were immunoreactive for epithelial markers (AE1/3; CK1, CAM 5.2, cytokeratin 7, and epithelial membrane antigen) (100%), calretinin (88%), vimentin (70%), androgen receptor (33%), and inhibin (30%, focal staining). No immunostaining was detected with cytokeratin 20, estrogen receptor, progesterone receptor, and monoclonal carcinoembryonic antigen. This staining profile is similar to that of mesonephric remnants and may be useful in the distinction of mesonephric carcinoma from mullerian endometrioid adenocarcinoma, with which it may be confused.

Endodermal sinus tumor of the mediastinum. Report of apparent cure in two patients with extensive disease.

Primary endodermal sinus tumor (EST) of the mediastinum has been regarded as a rare and rapidly fatal germ cell neoplasm. We describe two cases of extensive EST treated with a new high-dose sequential combination chemotherapy regimen (CISCA-VB) followed by radical surgical excision. They are alive at 11 and 20 months, respectively, postoperatively. These cases stand in marked contrast to previously reported series. A new approach to the management of this tumor is proposed.

Successful treatment of mediastinal germ cell/endodermal sinus tumors.

Three patients with endodermal sinus tumor of the mediastinum and two with endodermal sinus tumor mixed with other histologies were treated with CEBA (cisplatin, etoposide, bleomycin, and doxorubicin [Adriamycin]) and by cytoreductive surgery. Three patients treated with CEBA followed by surgery achieved a complete remission and remain free of disease at 36+, 35+, and 28+ months, respectively, from diagnosis. Two patients were treated initially with vinblastine, bleomycin, cisplatin, and surgery, and neither achieved a complete remission. Both then received CEBA, and one achieved a complete remission (duration 47 + months). Bone marrow toxicity from CEBA was severe, abrupt, and cumulative. All patients experienced neutropenic fever, anemia, and thrombocytopenia requiring platelet transfusions. The CEBA followed by vigorous cytoreductive surgery is effective treatment for germ cell tumors of the medastinum containing endodermal sinus tumor elements.

Malignant germ cell tumors of the mediastinum.

A review of 56 cases of primary malignant germ cell tumors of the mediastinum revealed that, as with benign teratomas, the tumors occurred in young adults (mean age 29 years) but that the sex distribution differed (86% male and 14% female). A single germ cell element was found in 37 (66%) of the tumors, and various combinations were present in the remaining 19 (34%). The tumors were classified among five recognized types of germ cell tissues. There were 24 seminomas (22 pure and two with mature teratomas), 17 embryonal carcinomas (nine pure and eight with mixtures), five teratomas, seven choriocarcinomas (three pure and four with mixtures), and three pure yolk sac tumors. Most (86%) of the patients were symptomatic at the initial examination, with chest pain, cough, and loss of weight being the most frequent presenting symptoms. The standard posteroanterior and lateral roentgenograms were the most helpful diagnostic tool, showing evidence of an anterior mediastinal mass in 53 patients. The diagnosis was established by surgical exploration of the mediastinum or by biopsy of a lymph node in 55 patients. Of the 55, 24 (43.6%) had complete resection of the tumor and 31 (56.4%) had incomplete resection or biopsy alone. The overall prognosis for mediastinal germ cell tumors is poor, partly because the tumors are far advanced at the time of diagnosis but also because some of the tumors that contain embryonal cell carcinoma, choriocarcinoma, and yolk sac elements are very aggressive. Factors that were prognostic in patients with seminoma--such as age, presence of the superior vena caval syndrome, lymphadenopathy, evidence of hilar disease on the chest roentgenogram, and resectability--were not predictive in patients with other types of malignant germ cell tumors. Although aggressive combination chemotherapy may represent a significant treatment modality for nonseminomatous mediastinal tumors, the present study spanned many years in which no chemotherapy was available. Patients in the later years of the study received combination chemotherapy with various treatment regimens. No conclusions concerning specific chemotherapy, therefore, can be derived from this study.

Second-look laparotomy in endodermal sinus tumor: a report of two patients with normal levels of alpha-fetoprotein and residual tumor at reexploration.

The role of second-look laparotomy in the management of patients with endodermal sinus tumor of the ovary is controversial. We report two women who converted to a normal alpha-fetoprotein (AFP) level during treatment with combination chemotherapy, yet were found to have residual endodermal sinus tumor at second-look laparotomy. In view of the limited experience with this rare disease, we continue to recommend second-look laparotomy for patients who have completed chemotherapy for endodermal sinus tumor of the ovary, regardless of the serum AFP level.

Pregnancy following removal and chemotherapy of ovarian endodermal sinus tumor.

The first reported case of successful pregnancy following unilateral adnexectomy and combination chemotherapy for nedodermal sinus tumor is presented. The rationale for conservative management of this virulent neoplasm is discussed.

Endodermal sinus tumor of the vulva in an infant.

An endodermal sinus tumor arising in the vulva of a 22-month-old infant is reported and the features of 3 previously recorded endodermal sinus tumors of the vulva are reviewed. Despite radical surgery, radiation therapy, and chemotherapy the tumor was fatal within 6 months. Only one of the 3 previously described patients with this type of vulvar tumor has survived more than 2 years. That tumor was the smallest reported, being the only one under 2 cm in diameter.

Cisplatin, vinblastine, and bleomycin combination chemotherapy in endodermal sinus tumor of the ovary.

The combination of cisplatin, vinblastine, and bleomycin was administered to 13 previously untreated patients with pure endodermal sinus tumor of the ovary. Twelve had unilateral salpingo-oophorectomy as initial surgery. Among 11 women whose alpha-fetoprotein (AFP) levels returned to normal after cisplatin, vinblastine, and bleomycin therapy, nine are alive with no evidence of disease after periods of 20 months to six years and two months. In two patients, AFP levels returned to normal only after the introduction of different combination chemotherapies. Hematologic toxicity was moderate. Pulmonary toxicity due to bleomycin occurred in two cases. All menstruating patients resumed normal menstrual function after the interruption of chemotherapy. Two patients delivered normal term infants.

Combination chemotherapy in the management of ovarian germ cell malignancies.

Twenty-four patients with ovarian germ cell malignancies received combination chemotherapy in the present series. Fifteen patients received intensive vincristine, actinomycin-D, and cyclophosphamide therapy for 12 to 18 courses, and 13 are alive and free of disease 34 to 86 months later. One patient with a stage III pure endodermal sinus tumor and one patient with a stage III mixed germ cell tumor composed predominantly of endodermal sinus tumor elements failed vincristine, actinomycin-D, and cyclophosphamide therapy, but each transiently responded to cis-diamminedichloroplatinum, vinblastine, and bleomycin therapy. Nine patients subsequently were treated on a new protocol that used the intensive vincristine, actinomycin-D, and cyclophosphamide regimen for five to six courses for all stage I ovarian germ cell malignancies and cis-diamminedichloroplatinum, vinblastine, and bleomycin therapy for advanced stage tumors containing endodermal sinus tumor. Each patient on the new protocol is alive and free of disease 14 to 26 months later. Short-term intensive vincristine, actinomycin-D, and cyclophosphamide therapy is recommended for all stage I ovarian germ cell malignancies requiring adjuvant chemotherapy. Preservation of ovarian and reproductive function is appropriate in the present group of patients. Vincristine, actinomycin-D, and cyclophosphamide therapy is also recommended for biomarker negative advanced stage ovarian germ cell malignancies. Cis-diamminedichloroplatinum, vinblastine, and bleomycin therapy is recommended for advanced stage biomarker positive ovarian germ cell malignancies. Serial alpha-fetoprotein (AFP) titers accurately reflect the status of endodermal sinus tumor elements and may be used as a guide to discontinue treatment for patients with pure endodermal sinus tumor malignancies, obviating the need for second-look surgery.

Mixed germ cell tumors of the ovary.

Forty-two patients with mixed germ cell tumors of the ovary who were treated at The University of Texas M. D. Anderson Hospital and Tumor Institute at Houston from 1944 through 1983 are retrospectively reviewed. The median age of these patients was 16 years. The most common symptom was abdominal pain, occurring in 38 patients (90%). Detailed analysis of clinical and pathologic features is presented. Twenty of 42 patients are alive and well. Current management of mixed germ cell tumors, including the roles of second-look laparotomy and monitoring of tumor markers, is discussed. Optimal treatment consists of surgery followed by combination chemotherapy.

Bacteremia in obstetrics and gynecology.

Surveillance of all episodes of bacteremia in the four major hospitals of a metropolitan area of 400,000 population between 1977 and 1981 revealed that bacteremia was documented in only 92 patients on obstetrics and gynecology services. Death was attributed to bacteremia in only four of these patients, three of whom had severe underlying diseases. These data confirm that death due to bacteremia in present-day obstetric and gynecology practice is extremely uncommon.

Surgery in ovarian cancer.

Ovarian cancer has the highest mortality of the cancers of the female genital tract. The signs and symptoms of ovarian cancer are either nonexistent or nonspecific. Women who are to undergo exploratory laparotomy without a definite diagnosis should have ovarian cancer considered as part of the differential diagnosis. Therefore, it is important that each surgeon know the current concepts of surgery for ovarian cancer. Documenting true extent of disease in those cases with limited ovarian cancer prevents the need for reexploration before adjuvant therapy. In those cases of disseminated disease, an attempt at tumor reduction should be undertaken, as reduction of tumor aggregates to less than 1.6 cm in greatest diameter improves prognosis. Surgery is the mainstay of therapy for ovarian cancer. Optimal initial surgery prevents the need for reexploration.