Pelvic sarcomas diagnosed as recurrence of Muellerian adenosarcoma of the uterus and of the mesosalpinx: clinical and histological study of two cases.
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Clinical and pathological features of two cases of pelvic sarcomas diagnosed as recurrence of Müllerian adenosarcoma arising in the uterus and in the adnexal region are presented.<BR>A 44-year-old woman (case 1), with a past history of total hysterectomy for uterine leiomyoma and "endometrial polyp" at 39 years of age, consulted us with a complaint of lower abdominal distension. Exploratory laparotomy revealed a large solid tumor within the pelvic cavity, which had no apparent relation to the adnexae or other abdominal organs. Extirpation of the tumor and bilateral salpingo-oophorectomy were performed. Two years after the operation, she died of recurrence of the tumor despite four reductive operations.<BR>A 50-year-old woman (case 2) was admitted to our hospital because of a lower abdominal mass, two years after total hysterectomy with bilateral salpingo-oophorectomy for microinvasive carcinoma of the cervix. Exploratory laparotomy revealed a large solid tumor within the pelvic cavity, which was independent from the gastrointestinal tracts and other abdominal organs. The tumor was excised, but she died ten month later because of its recurrence.<BR>Histologically, both tumors consisted of spindle-shaped mesenchymal cells with atypical nuclei and frequent mitotic figures, and were diagnosed as sarcomas in the pelvic cavity. Some tumor cells showed the feature of leiomyosarcoma in case 1.<BR>For identification of the origins of both pelvic sarcomas, the specimens obtained at the time of the previous surgeries were re-examined. Histologically, in case 1, the polypoid mass protruding into the uterine cavity was composed of benign epithelia associated with proliferation of the surrounding mesenchymal cells with atypical nuclei and myxomatous features. However, their mitotic figures were less than 2/10 high power fields (HPF). In case 2, the solid tumor arising in the left mesosalpinx contained histologically benign glandular structure and the surrounding mesenchymal component with markedly myxomatous features. Its mesenchymal cells had atypical nuclei, occasionally multinucleated, but showed very few mitotic figures. In addition, a lesion with similar histological features was found in the serosal surface of the uterine cervix. Accordingly, both tumors at their initial surgeries were diagnosed as Müllerian adenosarcoma, although both showed rare mitotic figures in the mesenchymal components.<BR>Collectively, the two cases of pelvic sarcomas were the result of local recurrence of Müllerian adenosarcoma of the uterus (case 1) and of the mesosalpinx (case 2), both of which had been initially considered to be benign. The fatal clinical course of our two patients with Müllerian adenosarcoma with infrequent mitotic figures implies that nuclear atypia of mesenchymal tumor cells and myxomatous change should be taken into account in the diagnosis of Müllerian adenosarcoma, in addition to the mitotic counts.
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