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Biphasic neoplasms with a benign to atypical epithelial component and a usually low-grade malignant stromal component have been reported in various sites, probably being best known for their occurrence in the uterine corpus (mullerian adenosarcoma). We report a tumor of this type that occurred in the testis of a 76-year-old man and, to our knowledge, is the first mesodermal adenosarcoma reported at this site. The patient had scrotal swelling for many years with a pronounced increase in the swelling over the past 2 years. A large complex solid-cystic testicular tumor was evident on ultrasound, and examination of a radical orchiectomy specimen showed a 6.5-cm mass. On microscopic examination, the neoplasm had a phyllodes-like appearance with bland cuboidal to flattened epithelium covering polypoid fronds, and lining glands and cysts. The stroma varied from cellular, particularly where it condensed around the glands and cysts, to hypocellular and hyalinized. It was immunoreactive for muscle specific actin, CD10, and to a lesser degree, desmin. This case expands the known sites where mesodermal adenosarcoma may occur. The histogenesis is speculative, but possible options are discussed.

A case of Mullerian adenosarcoma of the uterus is described. The patient, an 81-year old female, was treated in hospital for vaginal bleeding in 1993. She was diagnosed as having a tumour of the uterus. The patient underwent an abdominal hysterectomy with bilateral salpingo-oophorectomy. We found a polypoid intrauterine tumour which had led to a swelling of the cavum and cervical canal. Histologically a Mullerian adenosarcoma was diagnosed. The tumour consisted of a benign glandular component and an mesenchymal component in the form of a low differentiated pleomorphic rhabdomyosarcoma.

Mullerian adenosarcoma is a neoplasm composed of benign mullerian epithelium and a sarcomatous stroma. This tumor classically occurs in the endometrium of postmenopausal women and less frequently in the extrauterine genital tract. Rare cases of extragenital adenosarcoma have been reported. We present the case of a 23-year-old female who presented with an extragenital adenosarcoma arising in an endometriotic cyst in the pouch of Douglas. The patient was treated with local excision, chemotherapy and radiotherapy. At 2 years follow-up she was disease-free. The literature on extragenital adenosarcoma is reviewed. These tumors are clinically more aggressive than their uterine counterparts. Surgical excision is the initial treatment modality for these patients. Little information is available regarding the efficacy of adjuvant chemotherapy or radiotherapy. This is an area that requires further study.

Adenosarcoma of the uterus is a rare biphasic tumor containing benign glandular epithelial and malignant mesenchymal components. The tumor has been reported to be associated with antiestrogen therapy, particularly tamoxifen, but there have been a few case reports with MRI. We present two cases of MRI findings of uterine adenosarcoma after antiestrogen therapy, tamoxifen and toremifene in breast cancer patients. The tumor presents as a large polypoid mass occupying the endometrial cavity, and may protrude into the vagina. On MRI, the tumor typically shows solid components with scattered small cysts and heterogeneous enhancement. These findings are not significantly different from conventional adenosarcoma.

WOS: 000089600600014, PubMed ID: 11045336, Mullerian adenosarcoma of the uterus is a biphasic tumor exhibiting benign epithelial and malignant stromal component. This tumor may occasionally be associated with tamoxifen therapy which is used as an adjuvant drug for breast carcinoma. Reviewing the literature, we found only 12 adenosarcoma cases associated with tamoxifen therapy Thus, the clinical and pathological findings in a 58 years old postmenopausal woman who developed uterine adenosarcoma, following low dose tamoxifen therapy after 8 years, was discussed in this report.

A 71-year-old patient developed an uterine adenosarcoma two months after two years of tamoxifen adjuvant treatment for early breast cancer. After curettage for postmenopausal bleeding, the patient underwent a total abdominal hysterectomy with bilateral salpingo-oophorectomy and pelvic lymphadenectomy. Histologic examination revealed an adenosarcoma of the uterus without myometrial invasion or lymphatic spread. After four years of follow-up, there was no evidence of recurrent disease. The possible association between tamoxifen treatment and uterine sarcoma is discussed.

The clinical and pathologic features of 12 cervical adenosarcomas from the files of the Armed Forces Institute of Pathology are described. The patients ranged in age from 13 to 67 years (mean 37). The majority (58%) presented with abnormal bleeding. All tumors were located in the cervix and consisted of soft, tan, polypoid or papillary masses ranging in size from 1.5 to 4.5 cm. Microscopically, they showed a biphasic pattern with mesenchymal and epithelial components. There was a characteristic stromal condensation below the epithelial surface and around glandular structures. The cytologic atypia of stromal cells was 1+ in three, 2+ in five, and 3+ in four. The mitotic activity ranged from four to 28 (mean 7.0) mitotic figures per 10 high-power fields. One neoplasm contained cartilage and one striated muscle. Myometrial invasion was present in three. Treatment consisted of hysterectomy in nine patients and excisional biopsy in three. Two patients received radiotherapy; one before surgery and the other after hysterectomy. Two were treated with chemotherapy. Follow-up ranged from 9 months to 18.8 years. Nine patients were alive and well with no evidence of recurrent tumor at postoperative intervals of 0.8-18.8 years. One patient died 1 year after diagnosis with intraabdominal metastasis. One developed a recurrent tumor. This study demonstrates a favorable prognosis for patients with cervical adenosarcoma. Similar to patients with uterine adenosarcoma, prognosis is mostly affected by the presence of deep myometrial invasion.

A case of an extrauterine Müllerian adenosarcoma of the pelvic peritoneum is described. The patient, a 16-year-old girl, presented with abdominal distension and pain. At laparotomy, a tumor was found originating from the left pelvic side wall. Light microscopic examination revealed a Müllerian adenosarcoma. Postoperatively she was treated with polychemotherapy. Seventy months after initial presentation she is still free of tumor.

Mullerian adenosarcoma which is characterized by admixture of benign-appearing neoplastic glands and sarcomatous stroma, is an uncommon variant of malignant mixed mullerian tumor (MMMT). Classic treatment for this rare tumor is hysterectomy with bilateral salpingo-oophorectomy. Its prognosis is relatively poor, but better than MMMT. The efficacy of adjuvant postoperative radiotherapy and chemotherapy is debatable and appears not to be promising. We report a 62-year-old female with mullerian adenosarcoma diagnosed after hysterectomy with bilateral salpingo-oophorectomy, which had shown unique ultrasonographic feature like hydatidiform mole and had been diagnosed as fibroadenoma in endometrial curettage, with brief review of literatures.

Mullerian adenosarcoma is a rare tumor that usually arises in the uterus. Occasional cases arising in endometrial implants have been reported. We report the computed tomography (CT) findings of histologically proven mullerian adenosarcoma arising in perirectal endometriosis in a 46-year-old woman.

Background and objectives Mullerian adenosarcoma is an uncommon variant of mixed mesodermal tumour of the uterus. This is a case report of a 65 year old post-menopausal lady who presented with complaints of passing tissue fragments per vaginum for 2 days followed by spotting. On examination, a polypoid mass protruding through the cervix was seen which was biopsied. Following a preliminary histologic diagnosis of poorly differentiated sarcoma on the biopsy; the patient underwent total abdominal hysterectomy with bilateral salpingo-oopherectomy. Method The surgical specimen was formalin fixed and paraffin embedded. Haematoxylin and eosin stained sections were studied. Result and conclusion Histopathological examination of the polypoid mass revealed a tumour comprising of an admixture of benign endometrial glandular component with overgrowth of sarcomatous stromal component and heterologous elements. This may pose a problem in diagnosis due to its rarity, and hence its distinctive morphological features merit attention as described here. In view of the rarity of this tumor, it is mandatory to do extensive histologic sampling to identify areas of sarcomatous overgrowth before arriving at a diagnosis of mullerian adenosarcoma as the clinical course and management vary.

Background: Extrauterine adenosarcomata are thought to arise from pre-existing foci of endometriosis. In 1925 Sampson proposed three criteria for a definitive diagnosis of malignancy arising in endometriosis: (1) histological evidence of endometriosis in close proximity to the tumour; (2) no other identifiable primary site of malignancy; and (3) histological appearance of the tumour compatible with an origin in endometriosis. In 1953 Scott added a fourth criterion (4) requiring contiguous histological transition of benign endometriosis merging with the malignant component. Case Report: We report an unusual and unique case of extrauterine mullerian adenosarcoma which fulfills both Sampson’s and Scott’s criteria and review the literature on extrauterine adenosarcoma and their relationship with the extended mullerian system. We also demonstrate that lymph node “metastases” of adenosarcoma may arise from pre-existing endometriosis.

Mullerian adenosarcoma is a rare biphasic malignant neoplasm of the cervix characterized by an admixture of benign epithelial elements and a malignant sarcomatous stromal component, which may be either homologous or heterologous. An aggressive variant of adenosarcoma, mullerian adenosarcoma with sarcomatous overgrowth (MASO) is extremely rare, with only two such cases being reported in the English literature to date. In this report we present a case of MASO of uterine cervix with heterologous elements in a 15-year-old unmarried girl presenting with foul smelling menstrual bleeding and passage of fleshy masses. Because MASO with heterologous elements seems to appear at the earliest stages of reproductive lifespan in women, and have an uncertain malignant potential, gynecologists and pathologists should be aware and think about the possibility of this tumor.

A case of mullerian adenosarcoma of the uterus with sarcomatous overgrowth is described. The patient was also found to have bilateral polycystic ovaries. This 25 year-old white female presented with irregular vaginal bleeding and was diagnosed as having mullerian adenosarcoma with sarcomatous overgrowth by dilatation, curettage, and biopsy. Total abdominal hysterectomy, bilateral salpingo-oophorectomy with lymph node sampling were performed.

Extrauterine m¨ullerian adenosarcoma is a very rare tumor and it is characterized by a benign glandular component and a low-grade sarcomatous stromal component. These tumors have been reported to arise from ovarian or extraovarian endometriosis. However, there are scant reports on the MR findings of extrauterine mu ¨llerian adenosarcoma arising from deep pelvic endometriosis. We describe here a case of a large infiltrating extrauterine m¨ullerian adenosarcoma arising from recurrent deep pelvic endometriosis and we discuss its MR findings.

20533 Background: Uterine müllerian adenosarcoma is a rare tumor of postmenopausal women. An increase in its incidence with the use of tamoxifen therapy for breast carcinoma is reported in several studies. Methods: We report a case of 42 years multigravida, premenopaused woman with breast carcinoma, who presented - after four years of use of adjuvant tamoxifen - a synchronous liver, bone, and lung metastasis of breast cancer with müllerian adenosarcoma. Results: Immunohistochemical stains on uterine tumor for estrogen and progesterone receptors showed positivity for both epithelial and stromal cells, actin, desmin and proliferative index (MIB-1) showed positivity for stromal cells only. The patient underwent a hysterectomy followed by palliative chemotherapy. The patient deceased at 14 months after her relapse because of progressive disease (cerebral, bone, liver and lung metastasis). Conclusion: Our case is the only one reported in the literature with synchronous relapse of the breast adenocarcinoma and a Müllerian adenosarcoma. Also it's of the rare cases occurring in a pre-menopaused woman since all except two cases were postmenopausal. [Table: see text] No significant financial relationships to disclose.

Mullerian adenosarcoma has been described as an uncommon variant of Malignant Mixed Mullerian tumors (MMMTs) and a distinctive biphasic uterine tumor with benign proliferative glands and a sarcomatous stroma. It is considered that it is a tumor of low grade malignancy with relatively good prognosis, but the possibility of recurrence is still high and in occasional cases metastasis have been documented. We experienced one case that was diagnosed as Mullerian adenosarcoma of uterine corpus by pathologic biopsy obtained after surgery and report with the brief review of literatures.

Figure 1. A 34-year-old mother of two children had had menometrorrhagia, lethargy, and episodic abdominal pain for several months. A trial of oral contraceptives provided no relief. Dilation and curettage was performed; staining of the tissue with hematoxylin and eosin demonstrated mullerian adenosarcoma (Panel A, ×20). One month later, the patient was admitted for a total abdominal hysterectomy, bilateral salpingo-oophorectomy, and a staging procedure. Preoperative testing revealed an elevated serum level of the β subunit of chorionic gonadotropin, which is indicative of early pregnancy. After a consultation during which possible options were explained, the patient decided to proceed with the . . .

Mullerian adenosarcoma is an uncommon variant of uterine sarcoma. Twelve uterine adenosarcomas were diagnosed during a 42-month period at the Washington Hospital Center in Washington, DC. Based on estimated incidence data derived from the US Department of Defense beneficiary population, an estimated relative risk of 15.4 (95% confidence interval, 7.7-31.0) was calculated, indicating a significantly increased incidence of adenosarcoma in the population studied (p < 0.0000001). Among 10 patients who underwent hysterectomy, six (60%) of their tumors had sarcomatous overgrowth. In comparison with the previously reported proportion of adenosarcomas with sarcomatous overgrowth, approximately 16%, the proportion with sarcomatous overgrowth was significantly higher than expected (p < 0.01). Mullerian adenosarcoma with sarcomatous overgrowth was first described in 1989 and suggests that the cluster of adenosarcomas reported herein may be due in part to the current classification of some uterine tumors as adenosarcoma with sarcomatous overgrowth that previously would have been classified as other types of uterine sarcoma. Nonetheless, even after reviewing and updating the classification of all sarcomas diagnosed at the Washington Hospital Center from 1985 to 1998, the ratio of adenosarcomas to uterine adenocarcinomas during the 1994-1998 period was 4.7 times (p < 0.005) that of the 1985-1993 period, suggesting a more modest but real increase in the occurrence of this tumor. Correct classification of mullerian adenosarcomas with sarcomatous overgrowth is important because the limited available data suggest that the prognosis is notably worse than that for adenosarcomas without sarcomatous overgrowth.

Summary Extrauterine mesodermal (mullerian) adenosarcomas have only recently been described, and this is the first reported case from Australia. These tumours fall within the category of ‘common epithelial tumours’ in the World Health Organisation (W.H.O.) classification of ovarian tumours and comprise benign looking epithelial structures (glands, papillae) in association with sarcomatous stroma. They are thus distinct from malignant mesodermal mixed tumours in which both epithelial and stromal elements are cytologically malignant.

An unusual case of three concomitant primary uterine lesions is presented. The diagnostic and therapeutic problems are discussed.

The management of cervical adenosarcoma in a 14-year-old girl is described. The tumor had an exceptionally violent biological nature and did not respond to a variety of customary and unorthodox therapeutic measures. These included conventional intravenous chemotherapy, radiation therapy, surgery, intraarterial chemotherapy, colostomy, and peritoneovenous shunt for untreatable ascites. The patient died within 16 months of diagnosis. This is the fifth case of cervical adenosarcoma in the English literature. This tumor usually has a better prognosis and none of the previous four cases succumbed to the disease. The unusual virulence of the present case is discussed and the literature reviewed.

The clinicopathologic features of 4 cases of Müllerian adenosarcoma of the uterine body and 1 of the cervix are reported. They showed the characteristic pattern of benign epithelial growth within sarcomatous stroma. In the cervical tumor, malignant heterologous stromal cells were also present. All the patients are alive and well, confirming the good prognosis of this neoplasm. The authors think that the presence of a malignant heterologous component probably does not imply a worse prognosis.

Five extrauterine examples (three pelvic and two ovarian) of a distinctive mesodermal (mullerian) mixed tumor previously described to occur in the uterus and designated mullerian adenosarcoma are reported. The tumors were diagnosed during the fifth to eighth decades of life. They usually formed very large, partly cystic masses, which had occasionally spread to adjacent organs by the time of operation. On microscopic examination the tumors consisted of benign-appearing neoplastic glands lying in a sarcomatous stroma. The glands were lined by a variety of mullerian epithelial cell types, and the stromal element resembled endometrial stromal sarcoma. Three patients had malignant courses manifested by intra-abdominal recurrence, distant metastases, or both. This tumor should be clearly distinguished from the more common forms of mesodermal (mullerian) mixed tumor in which the epithelial as well as the stromal component is malignant and the prognosis is much more grave.

SummaryA Mullerian adenosarcoma of the uterine cervix in a 14 year old girl was studied by light and electron microscopy. The tumour was composed of a stromal sarcoma, benign glands of endometrial type and heterologous elements, such as rhabdomyoblasts, lipoblasts and mature striated muscle cells. On electron microscopic study stromal cells and rhabdomyoblasts in different stages of maturation comprised the major part of the tumour. The less aggressive behaviour of the Mullerian adenosarcoma in comparison to other uterine sarcomas and the benign appearing glandular element are distinctive features of this tumour. Stages of maturation of muscle cells and the origin of the benign element are discussed. An attempt was made to explain the less aggressive nature of the tumour.

Six cases of Stage I adenosarcoma of the uterus are reported. Although these neoplasms have generally been regarded as being of low malignant potential with little propensity for distant metastasis, our experience reveals some of them to be aggressive tumors with rapid clinical demise. Five (83%) patients demonstrated recurrence after initial surgery, three despite postoperative vaginal radium or external pelvic radiation. Four of the recurrences occurred in the pelvis and abdomen; the fifth was limited to the vagina. Three patients were dead of disease within three years of diagnosis, two of whom achieved clinical remission for at least one year to combination chemotherapy. One of the remaining three patients died from intercurrent disease without recurrence, and two are alive without disease after treatment for recurrence. Based on this limited experience, we believe these neoplasms should be regarded as potentially as malignant as their mixed Muellerian counterpart.A

A case of rhabdomyosarcomatous adenosarcoma of the uterine cervix is described and the literature reviewed. This variant of adenosarcoma does not appear to be more malignant than other adenosarcomas. It can and should be differentiated from embryonal rhabdomyosarcoma and malignant mixed müllerian tumor because of the different prognostic implications and therapeutic approaches.

A rare case of Mullerian adenosarcoma of the uterus is described, found during diagnostic curettage. Panhysterectomy was performed. These rare tumors have a low potential for malignancy and are characterized by benign epithelial and malignant stromal elements. They should be distinguished from the highly malignant mixed Mullerian tumors which contain, by definition, both carcinomatous and sarcomatous elements. The literature on the subject has been reviewed.

A case of mullerian adenosarcoma arising in an 82-year-old white female with rapid successive recurrence and death was studied with electron microscopy. The primary tumor in the uterus was typical mullerian adenosarcoma described by Clement and Scully. After a simple hysterectomy, however, a recurrent tumor appeared in the vagina in 5 months. The lesions were excised, and radiation therapy was given. The patient died 13 months after the initial diagnosis, reportedly with another recurrence of the tumor. Light microscopic and electron microscopic histology of the first recurrent nodules was consistent with fibrosarcoma.

This is a report of a transmission and scanning electron microscopic examination of a mullerian adenosarcoma. The neoplasm had the characteristic pattern of benign neoplastic glands with focal squamous metaplasia and a sarcomatous stroma. Ultrastructurally, both mesenchymal and epithelial elements were identified. The mesenchymal components were Ultrastructurally identical to those present in endometrial stromal sarcomas, including active collagen synthesis. The epithelial component resembled benign endometrial glands, and there was significant secretory activity as evidenced by prominent cytoplasmic glycogen collections.

A 52-year-old postmenopausal woman presented with signs and symptoms of acute pelvic inflammatory disease. On vaginal speculum examination, an infected polypoid tumor protruding from a dilated cervical canal was disclosed. While the patient was under extensive antibiotic treatment, uterine curettage was performed. This was followed a few days later by a total abdominal hysterectomy and bilateral salpingo-oophorectomy. Histopathological examination demonstrated the tumor to be Mullerian adenosarcoma of the uterine endometrium. The patient had postoperative vaginal vault irradiation, and more than 1 year later she is alive with no evidence of recurrent disease. Diagnosis and management of uterine Mullerian adenosarcoma is discussed.

This is a report of a Mullerian adenosarcoma of homologous type which arose from the right pelvic sidewall and presented as a pelvic hematoma in a 46-year-old woman. The tumor was locally aggressive and histologically similar to cases arising in the uterus reported earlier by Clement and Scully. Following her death from B. fragilis septicemia, the autopsy revealed widespread, occult hematogenous metastases composed entirely of the sarcomatous element of the tumor. The problems in diagnosis and management of a pelvic sidewall neoplasm are presented, and the methods of therapy of the homologous and heterologous forms of this new entity are reviewed and discussed in relation to their biologic behavior.