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Primary gastric adenosarcoma.

We describe the first case, to our knowledge, of a primary gastric adenosarcoma surgically resected from a 46-year-old white man. This biphasic neoplasm, well described in the female genital tract, was composed of cytologically benign tubular and cystic glands widely dispersed within a sarcomatous stromal component. Immunohistochemically, the epithelial elements were positive for cytokeratin, while the stromal elements were positive for vimentin and desmin. The clinical and pathologic features of pelvic adenosarcomas are described and compared with the tumor of the stomach. Although accurate prediction of the biologic behavior of this gastric neoplasm is difficult, the high mitotic rate in the sarcomatous component and the infiltration of the gastric serosal surface by the tumor were considered to be ominous prognostic features.

Humans↗

Epithelial-mesenchymal transformation of a newly established cell line from ovarian adenosarcoma by transforming growth factor-beta1.

We report on 2 cell lines (designated OVAK-I and OVAK-II) established from an adenosarcoma, a rare variant of a malignant Mullerian mixed tumor, of the ovary. OVAK-I was not tumorigenic in nude mice and showed no responsiveness to transforming growth factor (TGF)-beta1, whereas OVAK-II was tumorigenic and had various biological properties. Light- and electron-microscopic studies showed that the morphology of the cells was converted from an epithelial to a mesenchymal form by TGF-betaI. Furthermore, the protein and mRNA expressions of the epithelial marker carcinoembryonic antigen and the mesenchymal marker vimentin were down- and up-regulated, respectively, by TGF-beta1. These findings suggested that TGF-beta1 plays a role in epithelial-mesenchymal transformation and support the single-stem-cell theory which explains the origin of malignant Mullerian mixed tumors: that the epithelial and mesenchymal components of these tumors are of common stem-cell origin.

Adenosarcoma↗

The expression of epidermal growth factor receptor, HER-2/Neu, p53, and Ki-67 antigen in uterine malignant mixed mesodermal tumors and adenosarcoma.

Uterine malignant mixed mesodermal tumors (MMMT) are highly malignant tumors containing both malignant glands and stroma, while adenosarcomas (AS) are less aggressive tumors composed of malignant stroma and benign glands. Immunohistochemistry was used to grade overexpression of p53 protein, HER-2/neu protein, epidermal growth factor receptor (EGFR), and Ki-67 antigen in both the glands and stroma of tissue from 20 women with MMMT and 6 women with AS. EGFR was overexpressed in 2 AS and 9 MMMT, and was more commonly found in the sarcomatous component than the carcinomatous component in MMMT (P = 0.03). p53 was not found in any AS samples and was strongly present in 6 MMMT samples with a random distribution between the malignant components. HER-2/neu protein was not overexpressed in any AS or primary MMMT. Ki-67 antigen, a marker of cell proliferation, was found at higher levels in MMMT than AS samples (P = 0.03) and high Ki-67 antigen expression correlated with a decreased survival in patients with MMMT (P = 0.004). Independent characterization of oncogene proteins in the malignant components of these heterogeneous tumors may provide insight into the histogenesis and behavior of these malignancies.

Adenosarcoma↗

Adenosarcoma arising in hepatic endometriosis.

We report a case of adenosarcoma arising in hepatic endometriosis. Both CT and MR scans demonstrated a huge heterogeneous mass containing septated, thick-walled cystic lesions. After enlarged right hepatectomy, the patient was asymptomatic with no abnormalities at liver and abdominal CT scan at 2-year follow-up.

Adenosarcoma↗

A rare case of extrauterine adenosarcoma arising in endometriosis of the rectovaginal septum.

OBJECTIVE: To present a rare case of endometrial stromal sarcoma arising in endometriosis of the rectovaginal septum. DESIGN: Case report. SETTING: Academic tertiary referral center for endometriosis treatment. PATIENT(S): A 50-year-old woman with a history of endometriosis presented with catamenial rectal pain and deep dyspareunia. Imaging findings suggested new endometriotic lesions in the rectovaginal space. INTERVENTION(S): Total hysterectomy, salpingo-oophorectomy, and excision of the lesion in the rectovaginal septum were performed. Although extemporary pathology confirmed endometriosis, the final histologic diagnosis was extrauterine adenosarcoma in the rectovaginal septum. Two years later, recurrence of the malignancy occurred and was treated by resecting the new perirectal mass. Subsequent radiotherapy and chemotherapy were administered. MAIN OUTCOME MEASURE(S): Imaging findings at follow-up evaluation. RESULT(S): The patient was in good health for 2 years after the initial surgery, when she developed a new lesion at the site of the previous resection. The histologic appearance of the lesion was consistent with recurrence of the tumor. After postoperative therapy, the patient is now without evidence of disease. CONCLUSION(S): Malignant transformation of endometriosis should be considered in the differential diagnosis of a new pelvic lesion in a patient with a history of endometriosis.

Adenosarcoma↗

Uterine adenosarcoma in a boy with persistent müllerian duct syndrome: first reported case.

Persistent müllerian duct syndrome (PMDS) is a rare form of male pseudohermaphroditism. Bilateral fallopian tubes and a uterus occur with an otherwise normal male phenotype. Testicular neoplasm in PMDS has been reported, but malignant degeneration of the remnant female genitalia has only once been reported. We present imaging, surgical specimen, and surgical pathology of the first documented case of uterine adenosarcoma of a müllerian remnant in a 14-year-old boy with PMDS presenting with gross hematuria and increasing abdominal protuberance. The child died of metastatic disease after surgical resection.

Adenosarcoma↗

Late recurrence of uterine Mullerian adenosarcoma as heterologous sarcoma: Three recurrences in 8 months increasing in number and grade of sarcomatous components.

BACKGROUND: Uterine Mullerian adenosarcoma (UMA) is a rare and low-grade variant of mixed mesodermal tumor. UMA recurrences including heterologous sarcomatous elements are extremely rare, with only 3 cases reported thus far. CASE: We present a case of UMA recurring as heterologous sarcoma three times in 8 months for 9.4 years after primary treatment. The number and grade of sarcomatous components increased with each recurrence eventually resulting in loss of the patient. CONCLUSION: The presence of multiple sarcomatous elements of high histological grade in the recurrent tumor following UMA may indicate an unfavorable prognosis with common recurrences and fatal outcome. All UMA patients carry risk of late recurrence, therefore, they should receive long-term follow-up.

Adenosarcoma↗

Metanephric adenosarcoma in a young adult: morphologic, immunophenotypic, ultrastructural, and fluorescence in situ hybridization analyses: a case report and review of the literature.

Metanephric neoplasms are uncommon renal tumors that arise in both children and adults. They may be composed of small epithelial cells or benign stroma, or both, and are termed metanephric adenoma, metanephric stromal tumor, or metanephric adenofibroma, respectively. Thus far, these tumors have been known for their benign behavior. We present the case of a 21-year-old woman who developed a neoplasm composed of a renal epithelial component identical to metanephric adenoma combined with a malignant spindle cell sarcoma. The epithelial component was positive for pankeratin AE1/3, whereas the sarcomatous component was negative for epithelial markers and positive for vimentin, CD34, and CD117. No smooth muscle differentiation was apparent in the sarcoma by immunohistochemistry or ultrastructural analysis. By fluorescent in situ hybridization analysis of the sarcomatous component there was monosomy of the X chromosome, but no apparent variation from the normal diploid pattern for chromosomes 3, 7, 12, and 17. We conclude that the spectrum of metanephric neoplasia should be expanded to include malignant stromal variants, and we propose the term "metanephric adenosarcoma" for the present case.

Adenosarcoma↗

Immunohistochemical expression of CD10 antigen in uterine adenosarcoma.

Uterine adenosarcoma (UAS) is microscopically characterized by a biphasic growth pattern. By definition, the epithelial component is benign, whereas the stromal component typically has the appearance of a low-grade sarcoma, usually an endometrial stromal sarcoma. CD10 acts by reducing cellular response to peptide hormones and is currently regarded as a specific marker for endometrial stromal tumors. In this international multicenter study, we further explored CD10 immunoreactivity in 30 UASs. We encountered CD10 positivity of the sarcomatous component in 18/20 (90%) of UASs, in five of eight (63%) of UASs with sarcomatous overgrowth as well as in both cases of recurrent UAS. The epithelial component stained negative in all cases. These findings suggest that CD10 can be used to differentiate UAS from cellular leiomyoma, or in case endometrial stromal cells exhibit muscle differentiation. Furthermore, CD10 positivity in recurrent UAS might guide the pathologist toward an endometrial stromal origin.

Adenosarcoma↗

Müllerian adenosarcoma of the cervix: differential diagnosis, histogenesis and review of the literature.

A new case is reported of müllerian adenosarcoma, presenting as a 'benign cervical polyp' protruding through the vulva of a 44 year-old woman admitted with abnormal vaginal bleeding. This report emphasizes the importance of a careful examination of the stroma and special features of the entrapped glands in order to contribute to an earlier and proper diagnosis. The literature is reviewed and the probable histogenesis of this tumor and differential diagnosis with embryonal rhabdomysarcoma (sarcoma botryoides), adenofibroma, malignant mesodermal tumor and carcinosarcoma is discussed.

Adenosarcoma↗

Müllerian adenosarcoma of the uterus with sarcomatous overgrowth following tamoxifen treatment for breast cancer.

Müllerian adenosarcoma with sarcomatous overgrowth presented by a 52-year-old female patient after adjuvant tamoxifen treatment for breast carcinoma is described. The diagnosis was made on histological basis after curettage and complementary total hysterectomy with bilateral salpingo-oophorectomy. The immunohistochemical study showed high expression of estrogen receptors in the epithelial component of the lesion and irregularly positive findings in the stroma. The proliferative activity evaluated by Ki-67 immunoexpression was higher in the stroma than the epithelium. Some of the stromal cells showed rhabdomyoblastic differentiation. The association of tamoxifen use and development of mesenchymal neoplasms is discussed.

Adenosarcoma↗

Mullerian adenosarcoma of the uterine cervix.

Sarcomas in the uterine cervix are rare, the incidence being 0.5% to 1% of all cervical malignancies. This is a report of cervical mullerian adenosarcoma, which was encountered in a hysterectomy performed for prolapse. The tumor was composed of benign glandular elements and malignant stromal component, thus justifying its nomenclature. We wish to emphasize the distinctive morphological features of this rare cervical tumor.

Adenosarcoma↗

Gastrointestinal stromal tumor (GIST) with glandular component. A report of an unusual tumor resembling adenosarcoma.

A case of gastrointestinal stromal tumor (GIST) with an unusual glandular component is reported. The tumor was found in the gastric fundus of a 93-year-old woman. Histologically, the lesion showed a biphasic adenosarcoma-like structure. Typical low-grade spindle cell patterns of GIST were intermingled with numerous and partly cystic glands. The glandular epithelium had pyloric/foveolar-like appearance, with foci of intestinal metaplasia and low-grade dysplasia. The stromal component was immunoreactive for CD117 (c-kit) and CD34, and negative for myoid and neuroid markers. The ultrastructural examination found nondescript and undifferentiated spindle cells. The gastric mucosa and submucosa near the tumor contained a small area with features of gastritis cystica profunda, with glands similar to those present inside the tumor. Therefore, a collision of GIST and gastritis cystica profunda is suggested in the histogenesis of the lesion.

Adenosarcoma↗

Mullerian adenosarcoma of the uterine cervix.

Two cases of mullerian adenosarcoma with heterologous elements which occurred in uterine cervix and had a gross appearance of sarcoma botyroides are reported. Histologically, the tumours were composed of an admixture of benign appearing glands and a sarcomatous stroma, with heterologous elements consisting of skeletal muscle, bone and cartilage. No malignant epithetial components were observed. Indications are that, the prognosis of these tumours are much better than the usual malignant mixed mullerian tumours.

Adenosarcoma↗

[Adenosarcoma and other uterine sarcomas].

Sarcomas are tumors of conjunctive origin, rarely relating to the gynaecological sphere; among these, the adenosarcoma only represent one particular aspect. From one clinical case and from literature, the authors attempt to sum up the situation on the different clinical, para-clinical and therapeutic aspects of those bad-know neoplasia.

Adenosarcoma↗

Müllerian adenosarcoma of the uterine cervix with heterologous elements: a light and electron microscopic study.

A müllerian adenosarcoma with heterologous elements having the gross appearance of a sarcoma botryoides occurred in the uterine cervix of a 14-year-old girl. Histologically, the tumor was composed of an admixture of benign-appearing glands and a sarcomatous stroma, the latter containing areas of undifferentiated sarcoma and endometrial stromal sarcoma, as well as heterologous elements consisting of rhabdomyosarcoma and hyaline cartilage. No malignant epithelial component was observed. The distribution of benign müllerian-type glands throughout the tumor simulated a malignant mixed müllerian tumor in appearance. Ultrastructural studies of the stromal elements of the neoplasm suggest origin from primitive müllerian stroma, and the glands have features of primitive müllerian epithelium. This study supports the concept that the malignant mixed müllerian tumor and related müllerian sarcomas originate from a common undifferentiated multipotential müllerian stem cell.

Adolescent↗

Müllerian adenosarcoma of the uterus: an ultrastructural study of four cases.

Four cases of uterine müllerian adenosarcoma, a distinctive form of mixed müllerian tumor, were studied by light and electron microscopy. All tumors showed the characteristic histologic pattern of benign neoplastic glands within sarcomatous stroma. Ultrastructurally, both mesenchymal and epithelial cells were seen. The mesenchymal cells showed some features of endometrial stromal cells, including the presence of intracytoplasmic collagen fibers. The epithelial cells formed glands, which resembled benign endometrial glands and were separated from the stroma by a well-defined basal lamina. No transitional cells between the epithelial and mesenchymal cells were seen. The ultrastructural features of these tumors suggest that the sarcomatous portion is of endometrial stromal origin. The glandular portion may arise, along with the stroma, from multipotential stem cells, or the glands may be non-neoplastic entrapped endometrial glands stimulated by the stroma and thus appearing to form an integral part of the tumor.

Adult↗