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PubMed2002

Mesodermal (müllerian) adenosarcoma of the ovary: a clinicopathologic analysis of 40 cases and a review of the literature.

Forty cases of mesodermal adenosarcoma of the ovary occurred in women 30-84 years of age (mean 54 years). Abdominal discomfort and distension were the usual complaints. All the patients were treated with an oophorectomy, which was accompanied by a hysterectomy in 85%, a contralateral oophorectomy in 65%, and nonsurgical therapy in 28%. Tumor rupture occurred at or before the operation in 67% of the cases. Twenty-six tumors were stage I, 11 stage II, and 3 stage III. The tumors were unilateral in 97.5% of the cases and 5.5-50 cm (mean 14 cm) in greatest dimension; most of the tumors were predominantly solid but contained numerous small cysts. Microscopic examination revealed sarcomatous overgrowth in 12 tumors. Sex cord-like elements were present in six tumors (including four with sarcomatous overgrowth) and heterologous elements in five (including two with sarcomatous overgrowth). The highest mitotic index of the sarcomatous component was 1-25 (mean 6) mitotic figures per 10 high power fields. Only 6 of 26 women (23%) who were followed postoperatively for > or=5 years were free of tumor. In the other 20 patients recurrent tumor appeared at 0.4-6.6 years (mean 2.6 years) after operation as pure sarcoma (low grade or high grade) or adenosarcoma (with or without sarcomatous overgrowth). Eight women had additional recurrences, and four women had blood-borne metastases. One patient was alive at 15.7 years after the excision of pulmonary metastases. The 5-, 10-, and 15-year survival rates were 64%, 46%, and 30%, respectively. Age <53 years, tumor rupture, a high grade, and the presence of high-grade sarcomatous overgrowth appeared to be associated with recurrence or extraovarian spread. Ovarian adenosarcomas have a worse prognosis than uterine adenosarcomas, presumably because of the greater ease of peritoneal spread. Many of the tumors caused problems in differential diagnosis.

Adenosarcoma↗
PubMed1983

Uterine adenosarcoma: a clinicopathologic study of 11 cases with a reevaluation of histologic criteria.

Eleven biphasic uterine tumors with epithelial components and homologous stroma were reevaluated. Originally these were diagnosed as adenofibroma, adenosarcoma, carcinosarcoma, or mixtures thereof, but were now reclassified as adenosarcomas of which seven were "pure" and four mixed with foci of carcinosarcoma. Nine of the tumors arose in the endometrium and two in the endocervix. The mean patient's age was 55 years. The most common complaint was vaginal bleeding. Macroscopically these tumors presented as polypoid masses. The epithelial component consisted mainly of endometrial, endocervical, ciliated, and clear cells. Squamous metaplasia and focal hyperplasia were occasionally observed. Malignant epithelial change was only present in foci of carcinosarcoma. The stroma showed prominent cellular periglandular cuffs, occasionally round solid or perivascular nodules, and areas of focal or diffuse stromal hypercellularity. In all these areas stromal cells were atypical and/or pleomorphic. Stromal foam cells were seen in three cases. Mitotic activity was low ranging from one to three mitoses per 10 high power fields (HPF). Follow-up was negative exept in two cases with recurrence and abdominal metastases. It was concluded that stromal hypercellularity with atypism and pleomorphism in periglandular, perivascular location as well as of focal or diffuse nature, is characteristic of uterine adenosarcoma. Adenofibromas present a fibro-collagenous stroma lacking the crowded cellular areas. Mitotic activity is too variable to serve as a reliable diagnostic criterion. Uterine adenosarcomas are usually tumors of low grade malignancy but the lack of correlation between histologic appearance and biologic behaviour precludes prognostication in the individual patient.

Adult↗
PubMed1981

Endometrial adenosarcoma with pelvic involvement following uterine perforation.

An 85-year-old woman had a 7-year history of recurrent uterine adenosarcoma. One year after curettage-related uterine perforation, she developed a pelvic mass that was attached to the uterine serosa and was histologically identical to her endometrial lesions. The pelvic neoplasm probably resulted from implantation of tumor through the myometrial tear and is the first reported example of serosal adenosarcoma following myometrial perforation. The definition of sarcomatous stroma in Müllerian adenosarcoma, and thus its separation from adenofibroma, has not been delineated. A review of the literature indicates, however, that lesions recurring after hysterectomy have greater than three mitotic figures per 10 high-power fields. Incompletely excised neoplasms, treated by dilation and curettage only, often regrow, regardless of mitotic rate. Adenosarcoma may have a deceptively bland low-power pattern and must be differentiated from adenofibroma and benign polyps.

Aged↗
PubMed2006

Uterine adenosarcoma detected by Papanicolaou smear: a case report.

Adenosarcoma is a rare uterine biphasic tumor composed of benign epithelial elements and a sarcomatous stroma. Although it is well described histologically, its cytological features are rarely mentioned in the literature. We describe a case of uterine adenosarcoma that was first detected by Papanicolau (Pap) smear. Numerous crowded clusters of spindle cells were present within a bloody background, as well as a few smaller, dyscohesive groups with cells showing high N:C ratio and oval to round nuclei with coarse chromatin and small nucleoli. A few nuclear grooves were identified. Adenosarcomas are rare lesions but should be considered in the differential diagnosis when spindled cells are noted in a pap smear.

Adenosarcoma↗
PubMed2001

Uterine adenosarcoma with sarcomatous overgrowth versus uterine carcinosarcoma: comparison of treatment and survival.

OBJECTIVE: Uterine adenosarcoma with sarcomatous overgrowth (ASSO) is a rare variant of uterine sarcoma first described in 1989. This clinicopathologic study was undertaken to compare the treatment and survival of uterine adenosarcoma with sarcomatous overgrowth to that of uterine carcinosarcomas. METHODS: A review of uterine sarcomas diagnosed at Washington Hospital Center from January 1988 to December 1998 was performed. Records were reviewed for demographic data, surgical staging, primary and adjuvant therapy, metastatic site, disease recurrence, and survival. All pathology was reviewed and diagnosis confirmed. Statistical analysis included chi(2) test and Student's t test. Kaplan-Meier survival curves were plotted to estimate the median and 5-year survival times. The log-rank test was used to compare survival times. A P value <0.05 was considered significant. RESULTS: Sixty patients were diagnosed with uterine sarcoma at Washington Hospital Center. Of these, 33 (55%) were uterine carcinosarcomas, 11 (18%) ASSOs, 6 (10%) adenosarcomas, and 10 (17%) leiomyosarcomas. Of the patients diagnosed with uterine ASSO, 3 (27%) were stage I, 3 (27%) stage II, 1 (9%) stage III, and 4 (36%) stage IV. All 11 patients with uterine ASSO underwent total abdominal hysterectomy with bilateral salpingo-oophorectomy and tumor debulking. Postoperative adjuvant therapy included chemotherapy (n = 4), radiation (n = 4), combination radiation and chemotherapy (n = 1), and no adjuvant therapy (n = 2). The overall median survival time of patients with uterine ASSO was 13 months. Nine of eleven patients are dead of disease, and two patients (both with stage I) are alive without evidence of disease at 18 and 19 months. Thirty-three patients with carcinosarcoma were identified, with follow-up available on 29 patients. Of these, 10 (34%) were stage I, 6 (22%) stage II, 3 (10%) stage III, and 10 (34%) stage IV. Twenty-seven of the twenty-nine patients diagnosed with carcinosarcoma underwent surgical therapy to include total abdominal hysterectomy, bilateral salpingo-oophorectomy, staging and tumor debulking. Two patients died prior to treatment. Postoperative adjuvant therapy included chemotherapy (n = 9), radiation (n = 13), combination (n = 1), and no further therapy (n = 4). Twenty of the twenty-nine patients are dead of disease; there were nine surviving patients at the time of this report (stage I-5, stage II-3, stage III-1). The median survival of these patients was 31 months, with an overall 5-year survival of 22%. Comparison of the Kaplan-Meier survival curves using the log-rank test suggests a worse prognosis for uterine ASSO. However, this did not reach statistical significance (P = 0.0522). CONCLUSIONS: Patients diagnosed with uterine ASSO have a poor prognosis similar to that of carcinosarcoma. Management should include complete surgical staging. Additional therapy in the form of radiation, chemotherapy, or both has been reported; however, the superiority of one modality could not be determined from our data.

Adenosarcoma↗
PubMed2002

Use of medroxyprogesterone acetate in the treatment of Müllerian adenosarcoma: a case report.

BACKGROUND: Müllerianadenosarcoma is a rare pelvic malignancy that most commonly arises from the endometrium. These tumors are relatively insensitive to chemotherapy and radiation and are primarily treated by surgical resection. We report a case of müllerian adenosarcoma arising outside of the uterus from a background of endometriosis treated with a combination of surgical resection and medroxyprogesterone acetate. CASE: A 43-year-old woman with a history of endometriosis was diagnosed with advanced extrauterine müllerian adenosarcoma. After suboptimal tumor dubulking surgery she was treated with medroxyprogesterone acetate. Ten months postoperatively she remains without evidence of disease. CONCLUSION: Medroxyprogesterone acetate may be a useful drug in the treatment of advanced müllerian adenosarcoma.

Adenosarcoma↗
PubMed2000

Mullerian adenosarcoma of the uterus associated with tamoxifen therapy.

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.

Adenosarcoma↗
PubMed2004

Immunohistochemical determination of estrogen and progesterone receptor positivity in uterine adenosarcoma.

BACKGROUND: Given the paucity of data regarding hormone dependency, it was the purpose of this study to screen for the presence of estrogen and progesterone receptors in uterine adenosarcoma (UAS). METHODS: One hundred and five centers were asked to screen their files for uterine adenosarcomas. A immunohistochemical estrogen and progesterone receptor determination was performed. RESULTS: Twenty-eight primary UAS were stained, including one cervical adenosarcoma. Sarcomatous overgrowth could be observed in eight. Furthermore, two cases of recurrent UAS, one only consisting of endometrial stromal sarcoma, were stained. UAS lacking sarcomatous overgrowth showed estrogen receptor positivity in 17/20 (85%) and 16/20 (80%) in the epithelial and sarcomatous component, respectively. Progesterone positivity was observed in 13/20 (65%) and 12/20 (60%) in the epithelial and sarcomatous component, respectively. In 18/20 (90%) of the cases, either the estrogen or the progesterone receptor stained positive in the sarcomatous component. UAS with sarcomatous overgrowth showed estrogen receptor positivity in 4/8 (50%) and 0/8 (0%) in the epithelial and sarcomatous component, respectively. Progesterone positivity was observed in 2/8 (25%) and 1/8 (12%) in the epithelial and sarcomatous component, respectively. The stromal component of both recurrent cases stained moderately positive for estrogen receptor whereas progesterone receptor was considered negative. CONCLUSION: The observation that the sarcomatous component of UAS without sarcomatous overgrowth frequently expresses hormone receptors might be of significant clinical importance.

Adenosarcoma↗
PubMed2004

Primary vaginal adenosarcoma with sarcomatous overgrowth.

BACKGROUND: Primary vaginal adenosarcomas are extremely rare, and typical adenosarcomas are of low malignancy. However, aggressive forms with sarcomatous overgrowth have been reported, those appear to have a poor prognosis. CASE: A 52-year-old woman who had undergone prior surgery for uterine leiomyoma and an ovarian cyst (total abdominal hysterectomy and left salpingo-oophorectomy) presented 10 years later with a rapidly enlarging tumor arising from the vaginal cuff. Repetitive biopsy samples revealed a mixture of benign epithelial gland and malignant stromal components with periglandular stromal hypercellularity and sarcomatous overgrowth. A histological diagnosis of mullerian adenocarcinoma was made. The patient died from recurrent disease 9 months after surgery. CONCLUSION: Regardless of primary focus, adenosarcoma with sarcomatous overgrowth is associated with postoperative recurrence and a fatal outcome.

Adenosarcoma↗
PubMed2001

Adenosarcoma in a patient with vaginal endometriosis.

BACKGROUND: Adenosarcoma in a patient with extraovarian endometriosis is a rare event and can be easily overlooked. CASE: A woman with a history of endometriosis underwent multiple resections of a vaginal mass and medical treatment for presumed recurrent endometriosis. Eventually, a vaginal adenosarcoma was diagnosed. CONCLUSION: The possibility of adenosarcoma should be considered if an enlarging mass occurs at the site of extraovarian endometriosis.

Adenosarcoma↗
PubMed2003

Müllerian adenosarcoma of vagina arising in persistent endometriosis: report of a case and review of the literature.

BACKGROUND: Primary adenosarcoma arising in vaginal endometriosis poses a diagnostic challenge, especially in superficial vaginal biopsies. CASE: A 56-year-old woman, with a prior diagnosis of ovarian endometriosis, presented with a rapidly enlarging mass of the vaginal vault. Two prior biopsies were benign and showed endometriosis. The third vaginal biopsy revealed benign endometriotic glands cuffed by a cellular stroma with moderate cytologic atypia, a histological appearance diagnostic of Müllerian adenosarcoma. A 16-cm vaginal mass that had infiltrated the pelvic structures was resected. CONCLUSIONS: Close clinical follow-up of extrauterine endometriosis and clinical-pathologic correlation is necessary. Histological features such as cellular stromal cuffing around benign endometriotic glands are critical in arriving at a timely diagnosis of adenosarcoma in patients with persistent extrauterine endometriosis, even in superficial vaginal biopsies.

Adenosarcoma↗
PubMed2004

Molecular cytogenetic characterization of a case of Müllerian adenosarcoma.

Müllerian adenosarcoma is a distinctive type of mixed Müllerian tumor of the female genital tract. To our knowledge, no cytogenetic data have been documented on Müllerian adenosarcoma in the literature so far. We report here the chromosomal findings of a Müllerian adenosarcoma in a 15-year-old female. Cytogenetic and molecular cytogenetic analysis revealed a complex karyotype involving chromosomes 2, 8, 10, 13, 19, and 21. These numerical and structural abnormalities may be of etiologic significance. This report may highlight the potential value of molecular cytogenetic analysis in differential diagnosis of Müllerian tumors. More cases are warranted to further genetically characterize this type of neoplasm.

Abdominal Neoplasms↗
PubMed2005

[Adenosarcoma of the uterus: a case report].

Adenosarcoma of the uterus has two components: a benign epithelial proliferation and a stromal sarcoma. The purpose of this work was to report the clinical aspects and therapeutic modalities of an exceptional case of uterine adenosarcoma in a 15-year-old girl revealed by pelvic pain. Physical examination disclosed a polypoid formation protruding through the hymenal orifice. Histology of the mass confirmed the diagnosis of adenosarcoma of the uterus. The patient underwent hysterectomy and was given chemotherapy. This rare tumor (8% of uterine sarcomas) generally requires hysterectomy. Prognosis is relatively favorable.

Adenosarcoma↗
PubMed2000

Extrauterine Müllerian adenosarcoma of the peritoneum with an extensive rhabdomyosarcomatous element and a marked myxoid change.

A case of extrauterine Müllerian adenosarcoma of the peritoneum in a 20-year-old woman is reported. The tumor was widely based on the abdominopelvic wall and there were no unusual features in the genital organs. The cut surface of the tumor showed a marked gelatinous appearance. The tumor was composed of an admixture of benign Müllerian-type epithelium and sarcomatous stroma. The predominant element of the sarcomatous area was rhabdomyosarcoma, which showed a close resemblance to well-differentiated embryonal rhabdomyosarcoma. In another sarcomatous area, fibroblastic cells without myoblastic properties diffusely proliferated in a marked myxoid background with some collagen bundles. Both the mitotic count and Ki-67 proliferative index of these cells were lower than those of rhabdomyoblastic cells. On follow up, the patient was disease free for 1 year postoperatively, without any subsequent treatment. The present case indicates that extrauterine adenosarcoma can also show histological heterogeneity as do uterine adenosarcomas. The remarkable myxoid change of this tumor seemed to be more largely due to a fibromyxoid element than a rhabdomyosarcomatous element, and the coexistence of the former may be related to the less aggressive behavior of this tumor.

Adenosarcoma↗
PubMedDate not supplied

Uterine adenosarcoma with sarcomatous overgrowth: a case report with cytology of overgrown poorly differentiated sarcoma and immunohistochemical identification of epithelial microinvasion.

In this report, we describe a case of adenosarcoma of the uterine corpus with overgrown and poorly differentiated sarcoma. The patient was an 80-year-old Japanese woman with abnormal genital bleeding. The endocervical biopsy showed undifferentiated carcinoma, and a cytologic examination at that time revealed the presence of cell types ranging from dispersed spindle to round atypical cells, suggestive of mesenchymal origin. The resected uterus had a polypoid mass in the corpus of the uterus and a solid white mass with the appearance of frank malignancy in the endocervix. The phyllode pattern was evident in the polypoid lesion and a diagnosis of adenosarcoma with sarcomatous overgrowth was made. The epithelial component was atypical with occasional microinvasion, which mimicked rhabdomyoblasts, but was easily identified as microinvasion by positive staining for epithelial, but not myogenic, markers. This case stresses the importance of the cytologic smear in the identification of frank sarcoma in an adenosarcoma, and it furthermore highlights the usefulness of immunohistochemistry in the distinction of epithelial microinvasion from rhabdomyogenic differentiation.

Adenosarcoma↗
PubMed1995

Adenosarcoma of the uterine cervix: a clinicopathological study of 12 cases.

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.

Adenosarcoma↗
PubMed2006

Adenosarcoma arising in inguinal endometriosis.

BACKGROUND: Adenosarcoma arising in inguinal endometriosis is a rare clinical entity. CASE: A 47-year-old nulligravida presented with a persistent and enlarged right groin mass. Sixteen years earlier she had undergone total abdominal hysterectomy and bilateral salpingo-oophorectomy for endometriosis and was placed on estrogen therapy. Surgical en bloc excision of the 12 x 4 cm inguinal mass revealed adenosarcoma arising in endometriosis. CONCLUSION: Although a rare entity, adenosarcoma arising in inguinal endometriosis should be included in the differential diagnosis for those patients with a history of endometriosis and chronic unopposed estrogen therapy who present with a groin mass.

Adenosarcoma↗
PubMed2005

Mesodermal adenosarcoma of the testis.

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.

Actins↗