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PubMed2000

Management of uterine Müllerian adenosarcoma with extrauterine metastatic deposits.

OBJECTIVE: The aim of this study was to provide the management and outcome of three patients who presented with uterine Müllerian adenosarcoma associated with extrauterine metastases. METHODS: A retrospective study of three patients who were referred to our hospital was performed. One patient was referred because of vaginal metastatic deposits that were noted during investigations for primary infertility. The other two were referred because of abnormal vaginal bleeding; one of these had a large polyp protruding through her cervix into the vagina. RESULTS: In two patients the preoperative diagnosis and extent of their disease were known while in the third patient the diagnosis was only made postoperatively. All patients had a type II radical abdominal hysterectomy, bilateral salpingo-oophorectomy, and omentectomy. Two patients were given three cycles of neoadjuvant chemotherapy and pelvic irradiation over 12 weeks. Both of these patients had their diagnosis made preoperatively and the chemotherapy consisted of 240 mg/m(2) carboplatin and 80 mg/m(2) farmorubicin per cycle. The pelvic irradiation consisted of daily fractions of 1.8-Gy irradiation to a total of 45 Gy over the first 6 weeks. The other patient was given the same regime postoperatively. All patients are still alive and free of disease between 34 and 56 months. CONCLUSION: Radical surgery, chemotherapy, and irradiation provide a management option with seemingly favorable outcome for patients with uterine Müllerian adenosarcoma associated with extrauterine metastases.

Adenosarcoma↗
PubMed2002

Müllerian adenosarcoma of the cervix with heterologous elements: report of a case and review of the literature.

BACKGROUND: Müllerian adenosarcoma (MA) is a rare neoplasm composed of benign epithelial and malignant stromal components. Its location in the cervix and the presence of heterologous elements are extremely infrequent (to our knowledge, only 14 more cases have been reported in the English literature). We describe another case of MA of the uterine cervix with heterologous elements and review the clinical and pathological features of these tumors. CASE REPORT: A nulliparous 25-year-old woman was admitted to the hospital because of metrorrhagia and recurrent masses, dependent on the cervix, initially considered endocervical polyps. Microscopically, the last curettage performed revealed a tumor composed of two elements, epithelial and mesenchymal. The epithelial elements were benign endocervical type glands, and the mesenchymal were sarcomatous, containing minor foci of cartilage. A diagnosis of endocervical heterologous adenosarcoma was reached, and a total hysterectomy and bilateral salpingo-oophorectomy were performed. The patient is alive 2 years after the surgical procedure. Neither chemotherapy nor other adjuvant therapies have been administered, and she is clinically free of disease at the moment. CONCLUSION: Because MA with heterologous elements seems to appear at the earliest stages of the reproductive lifespan in women, commonly with a history of recurrent polyps, and because its malignant potential is uncertain, gynecologists and pathologists should be aware and think about the possibility of this tumor. Long-term follow-up is essential and so is the accumulation of individual cases to provide further experience with these unusual neoplasms.

Adenosarcoma↗
PubMed1995

Adenosarcomas originating from sites other than uterine endometrium.

We report three cases of adenosarcomas arising from extraendometrium of the uterus: one arising from the ovary, one from the paracolpium and one from the endocervix of the uterus. Microscopically, they consisted of an admixture of benign-appearing epithelial and mesenchymal components with hypercellularity and minimal atypia. Two of the tumors were initially misdiagnosed as endometriosis and one was diagnosed as adenofibroma. One patient had several recurrences and died 7 years after the initial laparotomy and another patient had sarcomatous overgrowth which invaded the muscular tissues of the large intestine. Thus it appears that adenosarcoma occasionally shows grave clinical behavior, despite the benign or low-grade appearance of its microscopic features. Problems of diagnosis and management of this tumor are discussed. An aggressive therapeutic approach including wide surgical excision is recommended even in questionable cases.

Adenosarcoma↗
PubMed2004

Endometriosis of the liver containing mullerian adenosarcoma: case report.

We present a case of a liver endometrioma in a postmenopausal woman. After failed management with leuprolide acetate, the mass was resected and contained focal areas of mullerian adenosarcoma. This is a rare case of mullerian adenosarcoma that appeared to arise within an endometrioma of the liver.

Adenosarcoma↗
PubMed2002

Laparoscopic management of 15 patients with infiltrating endometriosis of the bladder and a case of primary intravesical endometrioid adenosarcoma.

OBJECTIVE: To report laparoscopic management of 15 patients with infiltrative bladder wall endometriosis and to report a case of endometrioid adenosarcoma. DESIGN: Prospective chart review. SETTING: Referral center for endometriosis. PATIENT(S): Fifteen women with infiltrating endometriosis of the bladder. INTERVENTION(S): Laparoscopic segmental cystectomy and pathologic review of endometriotic bladder nodules in 15 patients. MAIN OUTCOME MEASURE(S): Location and characteristics of endometriotic bladder nodules. RESULT(S): Laparoscopic and cystoscopic evaluation confirmed that the endometriotic lesions were penetrating through the bladder wall. In 8 patients, the lesions were located in the dome of the bladder. In the remaining 7, the lesions were in the posterior wall, above the trigone. It was possible to treat all the lesions by performing a laparoscopic partial cystectomy. No intraoperative complications occurred. Deeply infiltrating endometriosis was confirmed on histologic evaluation in 14 cases. One patient was diagnosed with endometriosis on frozen section, but the final pathology revealed an adenosarcoma of the bladder. CONCLUSION(S): Surgical excision of deeply infiltrating endometriosis of the bladder wall can be performed laparoscopically and offers the benefit of a definitive pathologic diagnosis to rule out an occult malignancy.

Adenosarcoma↗
PubMed2003

A case of Müllerian adenosarcoma of the uterus treated with liposomal doxorubicin.

OBJECTIVE: We report on a case of uterine a denosarcoma responsive to treatment with liposomal doxorubicin (Doxil). METHODS: The clinical course, histopathology, and radiologic studies of the case were reviewed and are reported. RESULTS: A 69-year-old woman presented to our institution with dehydration and failure to thrive, and recurrent uterine adenosarcoma, 9 months after initial diagnosis. She had undergone total abdominal hysterectomy and bilateral salpingo-oophorectomy at the time of diagnosis, followed by two cycles of Ifosfamide and treatment with megestrol acetate. The patient presented to our institution for hospice care placement, severely dehydrated and with lethargy and with recurrent disease in the pelvis and vagina. She underwent treatment with six cycles of liposomal doxorubicin with a marked response. Given the development of Grade II hand-foot syndrome, liposomal doxorubicin was stopped and two cycles of carboplatin/paclitaxel chemotherapy were administered. Given the patient's disease progression on this regimen, surgical cytoreduction for localized recurrent disease was then performed. The patient developed disease recurrence after a 7-month disease-free interval and 2 years after initial diagnosis. Following another two cycles of liposomal doxorubicin, the patient underwent another cytoreductive procedure for recurrent disease. Two months later, the patient expired, 29 months after diagnosis and 20 months after initial treatment with liposomal doxorubicin chemotherapy. CONCLUSIONS: Liposomal doxorubicin appears to be active in the treatment of recurrent uterine adenosarcoma.

Adenosarcoma↗
PubMed1995

[Adenofibroma and adenosarcoma of the uterus in young women].

We report on two similar cases of young women with irregular menstrual bleeding. Curettage and biopsy revealed an adenofibroma of the uterus in case 1 (31 yrs) and an uterine adenosarcoma in case 2 (30 yrs). Hysterectomy was subsequently performed in both patients: histological examination of the hysterectomy specimen showed no remnants of the tumour in case 1. There was adenosarcoma tissue in the uterine cavity of case 2 but no myometrial infiltration. The difficulties of differential diagnosis of uterine soft tissue tumours in young women are discussed and the literature is reviewed.

Adenofibroma↗
PubMed1996

Mullerian adenosarcoma of the uterine corpus associated with tamoxifen therapy: a report of six cases and a review of tamoxifen-associated endometrial lesions.

Six consultation cases of mullerian adenosarcoma of the uterus were encountered in women who were receiving adjuvant tamoxifen therapy for carcinoma of the breast. To our knowledge, only one previous similar case has been reported. The women, who were 48-76 years of age, had received tamoxifen for periods of 6 months to 4 years (mean 2.7 years) in five of the cases; the duration of tamoxifen therapy in the sixth case is unknown. All of the tumors were polypoid endometrial masses that superficially invaded the myometrium in two cases. The microscopic appearance of the tumors was similar to that of previously described uterine mullerian adenosarcomas. These and other recent observations indicate that tamoxifen treatment may be complicated by uterine neoplasms other than endometrial adenocarcinoma. These findings also support previous observations that prolonged exogenous or endogenous hyperestrinism may lead to the development of mesenchymal and mixed epithelial-mesenchymal tumors of the uterus.

Adenosarcoma↗
PubMed2002

Primary peritoneal mullerian adenosarcoma with sarcomatous overgrowth associated with endometriosis: a case report.

We report a primary peritoneal mullerian adenosarcoma with sarcomatous overgrowth associated with endometriosis in a 50-year-old female. The tumor formed multiple peritoneal masses occupying the pelvis and subdiaphragmatic space. Histologically, the tumor was composed of benign-appearing mullerian glands surrounded by a sarcomatous stroma. In addition, one perisplenic mass was largely devoid of the epithelial component and was of higher grade than the remaining lesions. Multiple foci of endometriosis were associated with the pelvic masses. To our knowledge, primary peritoneal mullerian adenosarcoma with sarcomatous overgrowth associated with endometriosis has not been previously reported.

Adenosarcoma↗
PubMedDate not supplied

Mullerian adenosarcoma with sarcomatous overgrowth of the cervix presenting as cervical polyp: a case report and review of the literature.

An aggressive variant of adenosarcoma, mullerian adenosarcoma with sarcomatous overgrowth (MASO) in the cervix is extremely rare. This variant contains obvious, high-grade sarcoma in addition to a low-grade form. In this report, we describe a case of MASO of the uterine cervix and review the clinical and pathological features of these tumors. The patient was a 37-year-old woman with a cervical polypoid mass, which was morphologically considered as a benign endocervical polyp. Microscopically, polypoid cervical mass showed diffuse and dense malignant spindle cell proliferation around the benign endocervical glands and also an area of markedly anaplastic and pleomorphic spindle cell proliferation, so called, sarcomatous overgrowth. Total abdominal hysterectomy and bilateral salpingo-oophorectomy with pelvic lymph node dissection were performed. The patient has been followed-up and neither chemotherapy nor other adjuvant therapies have been administered. At present, she has been clinically free of disease for 9 months since she received surgery. It is extremely rare that MASO of the uterine cervix is presented in premenopausal woman. Gynecologists and pathologists should be aware of the difficulties associated with a delay in the diagnosis of MASO when the tumor is present as a benign looking cervical polyp.

Adenosarcoma↗
PubMedDate not supplied

Adenosarcoma of the uterus with extensive smooth muscle differentiation: ultrastructural study and review of the literature.

Four cases of Mullerian adenosarcoma were studied by light and electron microscopy and immunohistochemistry. All 4 cases showed the histologic characteristics of adenosarcoma with benign endometrial glands and a malignant stroma. Ultrastructurally, the epithelial component in all cases had the appearance of proliferative endometrial glands, and the malignant mesenchymal cells showed features of endometrial stroma. A distinct basal lamina separating glands from stroma was present. In addition, 2 of the cases showed extensive smooth muscle differentiation which was associated with sarcomatous overgrowth. The smooth muscle features were confirmed by immunohistochemistry. Multiple theories of the histogenesis of this tumor are discussed.

Adenosarcoma↗
PubMedDate not supplied

Clinicopathologic analysis of mullerian adenosarcoma: the M.D. Anderson Cancer Center experience.

The records of 41 patients diagnosed with adenosarcoma of the female genital tract between 1982 and 1996 were reviewed. The median age at diagnosis is 51 years (range, 14-84). The most common symptom is vaginal bleeding (71%). Clinical signs at presentation include pelvic mass (37%), uterine polyps (29%), and enlarged uterus (22%). In 71% of patients, the tumor originates from the uterus. Other sites include ovary (15%), pelvis (12%), cervix (2%). A history of thyroid cancer, benign ovarian cyst, and polycystic ovarian disease is found more frequently than expected in this patient population, whereas no relationship to endometriosis is observed. Surgery is the mainstay of treatment, but platin-based chemotherapy given upfront in inoperable patient has definite efficacy. An overall response rate of 92.5% was observed after primary therapy (surgery with or without radiotherapy, and/or chemotherapy), with a median survival of 48 months (range, 1-174). Thirty-eight percent of patients had recurrent disease. The median time to recurrence is 12 months (range, 5-132). Although 60% of patients with recurrence achieved a complete remission after treatment, only 1 (8%) is alive without disease, and 3 (22%), with disease at the time of this analysis. In our series, histologic sarcomatous overgrowth is a predictor of poor prognosis (p<0.03), however myometrial invasion and stage of disease seem to be of less prognostic significance. Adenosarcoma is a tumor with a fair prognosis. Most tumor can be cured with surgery, but recurrence carries a bad prognosis.

Adenosarcoma↗
PubMed2000

Mullerian adenosarcoma of the uterus: case report and review of literature.

Mullerian adenosarcoma--a variant of mullerian mixed mesodermal tumor of the uterus--is typically composed of benign but sometimes mildly atypical glandular epithelial elements admixed with malignant sarcomatous stroma. This rare tumor, which accounts for only about 8% of all uterine sarcomas, usually originates in the endometrium and grows as a polypoid mass within the endometrial cavity. The most prevailing presenting symptom is abnormal vaginal bleeding and the most common finding is a polypoid mass protruding through a dilated cervical canal. The case of a woman, who at age 62 presented with symptoms and signs of acute pelvic inflammatory disease and on vaginal examination an infected mullerian adenosarcoma protruding through a dilated cervical canal was discovered, is reported. Treatment consisted of extensive antibiotic treatment and surgery comprised of total abdominal hysterectomy and bilateral salpingo-oophorectomy followed by postoperative adjuvant pelvic radiotherapy. One year later, the patient is alive with no evidence of disease.

Adenosarcoma↗
PubMed2004

Adenosarcoma of the uterus: a case report.

Adenosarcomas of the uterus are very rarely observed and diagnosed. In this report, we present a case of a uterine adenosarcoma that was diagnosed in February 2002.

Adenosarcoma↗
PubMed2005

Uterine adenosarcoma diagnosed following hysteroscopic resection of an intrauterine tumour.

Uterine adenosarcoma is a mixed müllerian tumour consisting of a benign epithelial component and a malignant stromal component. It is a rare tumour that represents 8% of uterine sarcomas. We present a case of a 61-year-old woman who underwent a surgical hysteroscopy for postmenopausal metrorrhagia and thickened endometrium detected by ultrasonography. The pathologic diagnosis of the tumour removed by hysteroscopy was uterine adenosarcoma. The description of this case provides an opportunity to review the literature on uterine sarcomas diagnosed by surgical hysteroscopy.

Adenosarcoma↗
PubMed1997

Müllerian adenosarcoma of the uterus.

A case of adenosarcoma of the uterus in a 59-year-old woman is here reported. Adenosarcoma is a low malignant potential tumor with a benign glandular and a malignant stromal component. The treatment is usually hysterectomy with bilateral salpingo-oophorectomy. Debated is the usefulness of adjuvant chemotherapy, while radiation treatment is not beneficial. Long term follow-up is necessary for these patients because of high recurrence risk, mostly in cases with myometrial invasion.

Adenosarcoma↗
PubMed1978

Benign and low grade variants of mixed mesodermal tumor (adenosarcoma) of the ovary and adnexal region.

Eleven examples of a rare group of neoplasms composed of both epithelial and mesenchymal components are reported. Ten arose from the ovary and one arose separately in the para-ovarian region. The neoplasms are distinctive in that the stoma is more cellular than that of adenofibromas, but epithelial component is not malignant, as in carcinosarcoma and mixed mesodermal tumors, and the stoma is not sarcomatous in the low grade varieties. The 11 cases were highly variable in the cellularity and atypism of the stromal cells. The term, adenosarcoma, for these tumors is not acceptable because some were too low a grade to be regarded as sarcomas, and reports of the uterine counterpart disclose that some contain heterologous elements. For that reason, a term that will embrace the full spectrum of changes in the stroma--benign through sarcomatous--is needed. We propose that they be regarded as variants of mixed mesodermal tumor so that both the benign neoplasms and low grade sarcomas can be accommodated under one designation. Of the 11 cases, five lowest grade examples were all confined to the ovary and did not recur after surgical excision, but some of these were borderline in malignancy and probably would have progressed if untreated. Two of the 3 intermediate grade neoplasms extended beyond the ovary but were arrested by surgical excision. The 3 highest grade neoplasms were overly sarcomatous. One of these extended beyond the ovary but was arrested by combination chemotherapy. The para-ovarian adenosarcoma (also high grade) metastasized and proved fatal.

Adenofibroma↗
PubMed1986

Müllerian adenosarcoma with ovarian sex cord-like differentiation. A light- and electron-microscopic study.

A Müllerian adenosarcoma in which the sarcomatous element showed ovarian sex-cord like differentiation, occurred as a polypoid growth in the uterine cervix of a 53-year-old woman. The tumor was composed of an admixture of benign neoplastic glands and a sarcomatous stroma, the latter containing in addition to endometrial stromal sarcoma, nests of lipid-rich cells resembling ovarian sex-cord elements. This is the first report of such differentiation in Müllerian adenosarcoma. Origin from a focus of cervical endometriosis is postulated.

Cell Differentiation↗