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Reported here is a case of Müllerian adenosarcoma of the ovary which contained a primitive neuroectodermal tissue component within the stroma. The adenosarcoma coexisted with clear cell adenocarcinoma in an endometriotic cyst. The patient was a 33-year-old woman with a large unilocular endometriotic cyst of the right ovary. On the internal wall of the cyst, both a plaque-like protrusion with a papillary surface and a dome-like mural nodule were noted. The former exhibited features of Müllerian adenosarcoma, and the latter showed those of clear cell adenocarcinoma. In the deeper portion of adenosarcoma, teratoma-like tissue which contained various tissue components including primitive neuroectodermal tissue was found. The presence of primitive neuroectodermal tissue in the stroma of adenosarcoma suggested the diagnosis of 'adenosarcoma with neuroectodermal differentiation' ('teratoid adenosarcoma'), although the possibility of the incidental occurrence of an immature teratoma could not be completely excluded., (© 2011 The Authors. Pathology International © 2011 Japanese Society of Pathology and Blackwell Publishing Asia Pty Ltd.)

We describe a novel cystic renal tumor consisting of benign epithelial and malignant stromal components in a 56-year-old woman who was admitted to hospital with macroscopic hematuria. Enhanced computed tomography revealed a multilocular 3.4 × 2.7-cm tumor in the center of the left kidney. After total left nephrectomy, the excised tumor appeared extensively cystic with a well defined border on the cut surface. Histologically, the tumor was composed of biphasic a benign epithelial lining on tubules or cysts with a typically hobnailed appearance, and anaplastic sarcomatous stroma with frequent mitosis. Periepithelial cuffing of the sarcoma cells was evident without an epithelial-stromal transition. Carcinomatous nests, blastemic elements, ovarian-like stroma or differentiated mesenchyme were not evident in the stroma. The epithelial cells were reactive with cytokeratins, epithelial membrane antigen (EMA), vimentin and transducin-like enhancer protein 1 (TLE1). Stromal cells were reactive with vimentin, CD99 and TLE1, partly reactive with CD34 and CD10, and non-reactive with cytokeratins, EMA, Wilm's tumor protein (WT-1), estrogen receptor (ER), progesterone receptor (PgR), CD57, HMB45 or Bcl2. SYT-SSX fusion gene was not detected with reverse transcription polymerase chain reaction. Because these findings did not coincide with established descriptions of cystic renal neoplasms, we preferred the term, 'adenosarcoma'. This could become a new classification for adult cystic renal tumors., (© 2011 The Authors. Pathology International © 2011 Japanese Society of Pathology and Blackwell Publishing Asia Pty Ltd.)

We describe a case of adenosarcoma of the uterine corpus associated with ovarian thecoma in a 67-year-old woman. The patient underwent surgery under a diagnosis of ovarian carcinoma. The 110 x 70 mm-sized ovarian tumor was diagnosed as thecoma. The polypoid tumor of the uterine corpus which measured 30 x 15 mm was diagnosed as adenosarcoma. Cells of both epithelial and stromal elements of the adenosarcoma expressed estrogen receptors (determined by immunohistochemistry). These findings support the view that estrogen stimulation, including that by a pre-existing ovarian thecoma, may play a role in the development of mesenchymal and mixed epithelial / mesenchymal uterine tumors, including adenosarcoma.

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.

A case of adenosarcoma arising from the uterine cervix of a 55-year-old female who complained of vaginal discharge is reported. A polyp, 6 x 2 x 1.5 cm in size, identified in the cervical canal was clinically diagnosed as benign cervical polyp and resected. Histologically, the polyp was composed of benign epithelial components and sarcomatous stroma wherein periglandular hypercellularity and some mitoses including atypical ones were noted. Immunohistochemically, stromal cells were positive for muscle-type actins, desmin and estrogen receptor. Adenosarcoma is a rare, biphasic tumor of the uterus and usually presents as a polypoid mass in the endometrial cavity. When they arise from the cervix, adenosarcomas may be confused with benign cervical polyps clinically and pathologically. As the former often recurs, microscopic differentiation is crucial for further treatment.

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.